University of Groningen

Early intervention in infants at high risk for developmental motor disordersBlauw-Hospers, Cornill Henriëtte

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Early intervention in infants at high risk

for developmental motor disorders

RIJKSUNIVERSITEIT GRONINGEN

Early intervention in infants at high risk

for developmental motor disorders

Proefschrift

ter verkrijging van het doctoraat in de

Medische Wetenschappen

aan de Rijksuniversiteit Groningen

op gezag van de

Rector Magnificus, dr. F. Zwarts,

in het openbaar te verdedigen op

woensdag 1 december 2010

om 11.00 uur

door

Cornill Henriëtte Blauw-Hospers

geboren op 8 januari 1978

te Groningen

Promotor: Prof. dr. M. Hadders-Algra

Beoordelingscommissie: Prof. dr. J.G. Becher

Prof. dr. O.F. Brouwer

Prof. dr. P.J.M. Helders

Paranimfen: Victorine de Graaf-Peters

Joke de Boer

Contents

CHAPTER 1 Introduction

CHAPTER 2 The ‘Vroegtijdig Interventie Project’ (VIP)

CHAPTER 3 A systematic review of the effects of early intervention

on motor development

CHAPTER 4 Does early intervention in infants at high risk for a devel-

opmental motor disorder improve motor and cognitive

development?

CHAPTER 5 Development of a quantitative tool to assess the content

of physical therapy for infants

CHAPTER 6 Differences between the Family-centered Program

“Coping and Caring for Infants with Special Needs”

and Infant Treatment based on Principles of

NeuroDevelopmental Treatment

CHAPTER 7 A Randomized Controlled Trial on pediatric physical

therapy in infancy: from nightmare to dream?

CHAPTER 8 Additional results on outcome measures of the

VIP project

CHAPTER 9 General discussion

Summary

Samenvatting

Dankwoord

About the author

List of publications

Appendix

page 9

page 25

page 39

page 65

page 87

page 105

page 131

page 154

page 161

page 175

page 179

page 184

page 186

page 187

page 188

© C.H. Blauw-Hospers, 2010

Early intervention in infants at high risk for developmental motor disorders.

ISBN: 978 90 367 4606 9

Ontwerp + vormgeving: Mark van Houten, grafisch ontwerper

Druk: Grafische Industrie De Marne, Leens

All rights reserved. No part of this publication may be reproduces or transmitted in any form or by

any means, electronic or mechanical, including photocopy, recording, or any information storage

or retrieval system, without permission in writing from the author.

Niets uit deze uitgave mag worden verveelvoudigd en/of openbaar gemaakt door middel van druk,

fotokopie, microfilm of op welke andere wijze dan ook, zonder voorafgaande schriftelijke toestem-

ming van de uitgever.

9

C H a P t E r 1

I n t rod u c t Ion

10 11

In approximately 8% of all live births in the Netherlands preterm delivery occurs (CBS 2008). Ad-

vanced technology in neonatal intensive care over the past 20 years has increased the survival of

preterm and high-risk full term infants (Hack and Fanaroff 2000, Stoelhorst et al 2005). Follow-up

studies, however, have shown an increased incidence of motor disability later in life, ranging from

developmental coordination disorder to cerebral palsy (CP) (Hack and Fanaroff 2000, Marlow

2004, Stoelhorst et al 2005, Fawke 2007). To optimize developmental outcome of these infants

extensive early intervention programmes have been developed over the years. In this thesis I will

focus specifically on the effects of paediatric physical therapy programmes that have been applied

to infants up to 18 months corrected age who are at risk for a developmental motor disorder.

The aim of this thesis is to contribute to the body of knowledge about the effects of early

physiotherapeutic intervention for young infants who are considered to be at risk for a develop-

mental motor disorder. Existing programmes are reviewed and a new physiotherapeutic interven-

tion programme called ‘COPCA’ (COPing with and CAring for infants with special needs - a fam-

ily centred programme; Dirks and Hadders-Algra 2003) is evaluated. The goal of this evaluation is

twofold. First, the contents of COPCA are analysed and compared to the existing physiotherapeu-

tic approach. And second, the effects of the COPCA intervention on developmental outcome are

evaluated.

In this introductory chapter brief background information will be provided about the key subjects

in this thesis. In the following paragraphs information about early intervention, developmental

motor disorders of the infants in the studied cohort, and the main influences on paediatric physi-

cal therapy in the Netherlands will be provided. The chapter concludes with an outline of the

individual studies of this thesis.

Early Intervention

Young infants who are discovered to be at risk for developmental motor disorders are often re-

ferred to early intervention services. Early intervention (EI) consists of multidisciplinary services

provided to children from birth to 5 years of age (Shonkoff and Meisels 2000). The aims pursued

with EI are remedial or preventative in nature and comprise the following: 1) promote child

health and well-being, 2) enhance emerging competencies, 3) minimize developmental delays, 4)

remediate existing or emerging disabilities, 5) prevent functional deterioration and, 6) promote

adaptive parenting and overall family functioning (Shonkoff and Meisels 2000). These goals are

accomplished by providing individualized developmental, educational and therapeutic services for

children in conjunction with mutually planned support for their families. EI can be provided in

the hospital, in a rehabilitation centre, in the physical therapy practice, at home or in a combina-

tion of these four. Although EI may start at any moment between birth and 5 years of age it is

advocated to intervene as early as possible. It is recognized that only through early identification

and appropriate programming infants can reach their full potential. In a recent review on brain

development de Graaf-Peters and Hadders-Algra (2006) recommended that, based on develop-

mental processes, the best time window for EI in terms of active stimulation of the infant lies

between 40-44 weeks post menstrual age and 15 months postnatally. In this period, processes of

dendritic outgrowth and synapse formation are highly active which offer the best opportunities

to reconnect and find functional solutions than later periods (Kolb et al 2001, de Graaf-Peters and

Hadders-Algra 2006). Active stimulation consists of activities which induce active responses of

the infant, for example offering toys to stimulate head balancing exercises in prone position. De

Graaf-Peters and Hadders-Algra (2006) suggest also that EI prior to 40-44 weeks post menstrual

age should be restricted to forms which aim at mimicking the intrauterine environment in order

to reduce stress. Other advantages of intervening early are possible beneficial effects on the family.

From the literature it is known that families who encounter problems with their infant’s develop-

ment experience higher levels of stress (Singer et al 1999, King et al 2004, Muller-Nix et al 2004,

Butcher et al 2008). EI can help to minimize the impact of developmental problems on the family.

Summarizing, the research field of EI is very broad. It covers a wide age span and a whole range

of services. Therefore in this thesis the term early intervention is limited to paediatric physical

therapy for infants up to 18 months corrected age who are at risk for a developmental motor disorder.

Developmental motor disorders

Cerebral palsy and developmental coordination disorder are two important forms of developmen-

tal motor disorders. Cerebral Palsy (CP) is defined as “a group of disorders of the development of

movement and posture causing activity limitation, which are attributed to non-progressive distur-

bances that occurred in the developing foetal or infant brain. The motor disorders of cerebral

palsy are often accompanied by disturbances of sensation, perception, cognition, communication,

behaviour, by epilepsy and by secondary musculoskeletal problems” (Bax et al 2005, Rosenbaum

et al 2007). The prevalence of CP in the Netherlands is around 1 or 2 children per 1000 live births

with an incidence which is higher in males than in females (1.33:1) (Wichers et al 2005). CP is the

result of an injury or abnormal development of the brain which may occur during pregnancy,

birth or within the first two to three years of life. In many cases, the exact cause of CP is unknown

(Rosenbaum 2003). Currently, problems occurring during intrauterine development, congenital

disorders, hypoxic-ischemic insults, and preterm birth are considered to account for the major-

ity of the cases with CP (Moster et al 2001, Jones et al 2007). Infants born prematurely have an

increased risk for intraventricular hemorrhage (IVH) or periventricular leukomalacia (PVL) result-

ing in injury to the periventricular white matter of the brain. PVL is a good predictor for CP, with

CP developing in 80% to 85% of the infants (Perlman et al 1996, Jones et al 2007). All children with

CP suffer from some degree of motor impairment which in most cases manifests itself before 18

months of age and persists throughout the lifespan. During early infancy CP cannot be diagnosed,

because in many cases the characteristic features belonging to CP have not manifested them-

selves. The way CP affects each individual will vary widely. Characteristics may include a lack of

coordination, spasticity, involuntary movements, muscle weakness, increased tendon reflexes, dif-

ferent walking patterns (i.e. scissors gait, toe walking, wide-based gait), dysarthria, difficulty with

12 13

gross and fine motor skills, and abnormal perception and sensation.

The clinical picture of CP evolves with time, development, learning, training, therapies, and

other factors (Bower and McLellan 1994, Bax et al 2005). Therapy therefore focuses on helping the

individual to maximize his or her potential (Scrutton 1984). In general it is believed that the earlier

treatment starts the better chance children have of learning new ways to accomplish challenging

daily tasks. While this seems intuitively obvious, strong scientific support is lacking to support

this assumption (Damiano 2006).

Developmental coordination disorder (DCD) denotes children whose performance in daily activi-

ties that require motor coordination is substantially below what can be expected given the child’s

chronological age and intellectual ability, in the absence of other neurological disorders (APA;

DSM-IV; 2000). The disturbance in the performance of daily activities interferes with academic

achievement or activities of daily living. Estimates of the prevalence of DCD range from 5% to

15% in the primary school population (Wilson 2005). Most studies on children with DCD report

a higher prevalence in boys than girls, but the actual sex ratio is variable and depends on the

method used to identify children (Missiuna et al 2006).

Children with DCD not only differ in the motor skills they experience problems with, or the

underlying processes that are causing the deficit, but also in the extent to which they suffer from

co-morbidities such as attention, behavioural or emotional problems (Dewey et al 2002, Gillberg

and Kadesjö 2003). Studies on the underlying processes have shown that children with DCD can

have deficits in many areas of performance, including motor planning, motor programming, pos-

tural control, proprioceptive or kinesthetic deficits, and visual spatial processing (Niemeijer 2007).

About half of children with DCD will not outgrow their motor problems (Losse et al 1991, Cantell

et al 2003).

DCD is difficult to diagnose before school-age. Clumsy motor behaviour becomes more evi-

dent with increasing difficulty of motor tasks. Research has shown that a relation exists between

the presence of neurological dysfunction in infancy and DCD (Jongmans et al 1997, Hadders-Algra

2003). A few studies have shown that early identification (i.e. before school-age), effective interven-

tion and vocational counselling are important for children diagnosed with DCD to diminish the

negative experiences that can affect their later academic and social life (Cantell et al 2003, Mis-

siuna et al 2003).

advances in paediatric physical therapy

In the early 1900s European physical therapists were trained in manipulation by a London in-

ternist and orthopaedic surgeon Dr. James Cyriax. CP at this time was considered to be a disease

which they tried to cure with orthopaedic surgery. Over the years many different intervention

programmes have been developed for young children with CP varying from different types of

physical therapy and occupational therapy to the use of drug such as baclofen or botox, orthotic

devices, etcetera. The variety in approaches might result from the diversity in effect that CP has

on the daily functioning of the child (Mayston 2004). In this introductory chapter a brief overview

of the main influences on paediatric physical therapy for infants with or at risk for CP is provided.

In the Netherlands the main influences come from Neurodevelopmental Treatment (NDT), Vojta,

Sensory Integration Therapy, and more recently from functional therapy. The intervention pro-

grammes differ in their treatment methods, but all aim at ‘leading the children with cerebral palsy

… towards the greatest degree of independence possible, and so to prepare them for as normal as

an adolescent and adult life as can be achieved. This is the aim of all schools of treatment’ (Bobath

and Bobath 1984, p.6).

NeuroDevelopmental Treatment

The NDT approach was developed by Berta and Karl Bobath in the 1940s. The basic assumption of

NDT was that sensorimotor problems in CP arise from central nervous system dysfunction, which

interferes with the establishment of normal motor development. Inhibition of spasticity and

facilitation of normal posture and motor behaviour were the main aims of treatment, which the

Bobaths tried to accomplish by tone influencing patterns with the use of various therapeutic han-

dling techniques (Bobath and Bobath 1984, Mayston 2001). The normal developmental sequence

was advocated as a framework for treatment (Bly 1991, Howle 2002). In the sixties Elizabeth Köng

and Mary Quinton started to adapt and develop the techniques for early treatment of babies based

on the Bobath concept (Köng 1966, Quinton and Nelson 2002).

During the years the NDT approach evolved from a practice-based concept to a more theory-

based and partly evidence-based approach (Bly 1991, Palmer 1997, Mayston 2000, Howle 2002).

This means that based on new theoretical constructs and practical knowledge NDT gradually

evolved and caused a shift in goals, treatment techniques and treatment application (Howle 2002).

The original goal ‘inhibition of spasticity and facilitation of normal posture and motor behaviour’

to give the child the opportunity to move more normally nowadays has shifted to ‘optimize func-

tion’ (Howle 2002). This implies that NDT intervention aims to prepare and guide the children

towards their greatest possible independence in everyday functioning (Bobath and Bobath 1984).

Based on experience and new insights in neurosciences the emphasis on certain treatment

techniques changed. In the early years specific postures were imposed on the child with the aim

of achieving a normalization of muscle tone. The ‘reflex-inhibiting’ postures and tone influenc-

ing patterns indeed did reduce spasticity during treatment, but lacked carry-over effects into

movement and function (Mayston 2001, Howle 2002). Next, the Bobaths promoted the use of ‘key

points of control’, i.e. special combinations of hand placement on the body that allowed the thera-

pist to control the child’s sensorimotor input and motor output. These handling techniques had

the dual aim of inhibiting abnormal movement patterns and facilitation of normal movements

(Bobath and Bobath 1984, Butler and Darrah 2001, Howle 2002). Moreover, therapeutic handling

from key points of control made it possible for the child to - systematically and increasingly - take

over control of his/her own movement.

Over the years therapists gradually became more aware that their treatment had not automat-

14 15

ically carried over into activities of daily life. As a consequence, preparations for functional tasks

currently are carried out in settings where children live, play and learn (Bobath and Bobath 1984,

Butler and Darrah 2001, Howle 2002). The principles for the NDT baby treatment (Bly 1999) aim

at helping the infant in its development by the use of sensorimotor experiences (handling) and in-

structing the caregivers. In this regard caregivers play a specific role in NDT treatment. They have

a role as co-therapist (Finnie 1996). Through guiding and training of caregivers, the therapist tries

to establish carry-over from treatment into activities of daily life (Bobath and Bobath 1984). The

hands of the therapist facilitate, assist or control the infants’ posture and movements until the

infant can take over. The treatment consists of a sequence of developing, kinesiological, sensory

and environmental factors.

Treatment techniques

Tone influencing patterns are the primary intervention strategy of NDT that are achieved through

various therapeutic handling techniques. The use of a tone influencing pattern inhibits abnormal

activity, while at the same time the way is paved for more normal movements (e.g. facilitation)

(Howle 2002). However, in 1965 the Bobaths already acknowledged that: “unless you stimulate or

activate your patient in a way in which new activities are possible, you have done nothing at all. So

the tone influencing patterns as such are only the very first step in treatment, though they are very

important” (Mayston 2005). Thus, during NDT treatment inhibition and facilitation always act as

interactive partners in order to prepare the body for the performance of functional tasks. Further-

more, therapeutic handling techniques allow the therapist to provide sensorimotor experience, to

feel responses to changes in posture and movement, to gain control of muscle tone, to influence

the alignment of the body, to direct the attention of the child and to recognize the moment when

the child can become independent of assistance (Howle 2002). Therapeutic handling consists of

the following interactive components:

1) Key points of control are predefined parts of the body on which the hands of the therapist are

placed to influence directly the area beneath the hands of the therapist and other parts of the

body. The key points of control allow the therapist to inhibit abnormal movement patterns

and to facilitate normal movements simultaneously (Bobath and Bobath 1984). Key points are

located all over the body and are used interchangeably and in combinations. During treatment

there is a shift in the use of key points from proximal (e.g. shoulder, pelvic girdle) to distal (e.g.

extremities) to give the child the opportunity to gradually gain control over his movements.

2) Facilitation is the strategy of therapeutic handling that makes a posture or movement more

likely to occur. It can modify postural control by increasing degrees of freedom, supporting a

body segment, or activating the postural system. Placing the child in an imposed posture creates

a possibility to promote the occurrence of normal movement patterns (Howle 2002) (p.255-256).

3) Inhibition originally referred to reducing tone and abnormal reflex activity resulting from dys-

function in the central nervous system. Currently, inhibition refers to the reduction of specific

underlying impairments that interfere with function (Howle 2002) (p.257). Specific goals are to

gain control of muscle tone, to decrease excessive co-activation patterns and to prevent the oc-

currence of atypical postures.

As NDT is the most widely applied method in the treatment of infants with or at risk for develop-

mental disorders, many studies are undertaken to demonstrate its effectiveness. In three recent

reviews (Butler and Darrah 2001, Blauw-Hospers and Hadders-Algra 2005, Spittle et al 2007)

attention was paid to the effectiveness of NDT. All three concluded that there was no advantage

to NDT over other alternatives to which it was compared. No clear evidence was found that NDT

was beneficial for motor development or functional motor activities. Also other potential benefits

such as the enhancement of cognitive development or improved caregiver-infant interaction could

not be demonstrated. Possible explanations that are provided for the scarceness of evidence are

for instance the ongoing change of the constructs of NDT over time, the skill level of the therapist

and the impossibility to deliver treatment in a standardized manner (Butler and Darrah 2001).

While for research purposes the treatment setting can be standardized, the child with CP and

his family can never be. Moreover, CP is a highly heterogeneous condition which is complicated

by ongoing change in the children due to processes of growth and maturation. The change of the

NDT constructs over time also includes that techniques of NDT were more often used together

with techniques from other approaches that gained influence in the treatment of children with

developmental disorders, like for example the Vojta concept and sensory integration therapy.

Treatment according to Vojta

The Vojta concept, also called reflex locomotion, was developed during the 1950s by Vaclav Vojta

based on empirical observations. It was implemented in the Netherlands in the 1970s and is still

applied, especially in the southern part of the country. The method is primarily considered to be

a facilitatory approach that can be applied to all neuromuscular conditions, not just CP. Goals of

reflex locomotion are: 1) to facilitate the automatic control of the body position, 2) to facilitate the

uprighting mechanism of the body, and 3) to stimulate coordinated muscle activity. These skills

are disturbed in children with brain lesions or developmental motor disorders.

The concept is based on the observation that through specific peripheral stimulation nor-

mal motor reactions throughout the body can be elicited when the patient is placed in a specific

position. Vojta believed that the persistence of newborn reflexes in a child with CP interferes

with normal postural development. It is assumed that the activation of newborn reflexes through

stimulation of the global reflex pattern facilitates the development of reflex locomotion (Vojta

1984, Stanger and Oresic 2003, Mayston 2004).

Treatment techniques

The Vojta concept actually consists of two parts: clinical evaluation and treatment. Before the

physiotherapist can start with the reflex locomotion treatment, he can use the Vojta concept as

16 17

a clinical evaluation to define the goals for the treatment. The clinical evaluation is divided into

three parts. The first part is the study of the automatic postural reactivity. To discover potential

problems in the organization of automatic responses by the CNS, reactions to sudden postural

changes of the body, i.e. disturbances of the equilibrium position, are tested. This helps to identify

the degree of development at time of the examination. The second part consists of the kinesio-

logic analysis of spontaneous motor function. The placement of the extremities at specific angles

is important to maintain the equilibrium of the body. The third part is reflexology. A series of

reflexes completes the examination to refine the immediate evaluation but also to specify the

prognosis (Vojta 1984).

In the practical application reflex locomotion utilises two so-called coordination complexes:

the reflex creeping and rolling. Vojta identified a number of reflex points which can be used

to stimulate the human body to crawl and turn. From several start positions (supine, prone or

side-lying) the flow of motor responses is provoked by giving pressure on specific body parts, the

so-called ‘zones’. During the reflex locomotion it is essential that the placement of the extremities

at specific angels is optimal. The motor responses that arise during the application of the reflex

locomotion are called ‘global patterns’. Global implies that skeletal muscles throughout the body

are activated in a coordinated manner and that the central nervous system (CNS) is addressed at

all it’s regulatory levels (Vojta 1976).

Caregivers play a specific role in treatment according to Vojta. They are thoroughly instructed

to carry out the patterns of reflex locomotion. Optimally, the therapy should be carried out 4

times a day. The child is not actively involved in the treatment.

Vojta states that the best results of reflexlocomotion are achieved in babyhood when the plastic-

ity of the brain is greatest. He claimed that pathological motor patterns are already present but

not yet fixated and that the opportunity to gain access to normal movement patterns is merely

blocked (Vojta 1976, 1984).

The effect of treatment according to Vojta is evaluated in only a few studies. Blauw-Hospers

and Hadders-Algra (2005; see Chapter 3) showed that there was no evidence that treatment ac-

cording to Vojta supports the development of infants at high risk for developmental motor disor-

ders. The Vojta approach has been criticized for the emotional stress it may produce in child and

family (d’Avignon et al 1981).

Sensory Integration Therapy

Sensory integration therapy was developed during the 1960s by Jean Ayres. She was interested

in the way in which sensory processing and motor planning disorders interfere with activities of

daily living and learning. Sensory integration is the process by which people register, modulate,

and discriminate sensations received through the sensory systems to produce purposeful, adap-

tive behaviours in response to the environment (Ayres 2005). Sensory integration therapy is based

on the assumption that learning is dependent on the ability to take in sensory information from

the environment, to process and integrate the sensory inputs within the central nervous system

and to use this information for the planning and production of organized behaviour (Ayres 1972,

Mayston 2004). Sensory integration focuses on the five sensory areas: vestibular, tactile, pro-

prioceptive, visual and auditory information. The goal of intervention is to improve the ability

to process and integrate sensory information and to provide a basis for improved independence

and participation in daily life activities, play, and school tasks (Schaaf and Miller 2005). Although

originally sensory integration therapy was developed to treat children with learning disabilities,

Ayres recognized the utility of the theory for other clinical populations (Schaaf and Miller 2005).

As a significant amount of the children with CP also experience sensory impairments, sensory

integration may assist the child during the intake and processing of sensory information and thus

enhance function.

The theory of Sensory Integration is based on principles from developmental psychology,

education, neuroscience, and occupational therapy. It entails four main principles that are: 1)

sensorimotor development is an important substrate for learning; 2) the interaction of the indi-

vidual with the environment shapes brain development; 3) the nervous system is capable of change

(plasticity); and 4) meaningful sensory–motor activity is a powerful mediator of plasticity (Schaaf

and Miller 2005).

Treatment techniques

The therapy is driven by four main principles: 1) Just right challenge (the child must be able to

meet the challenges through playful activities), 2) Adaptive response (the child adapts behaviour

to meet the challenges presented), 3) Active engagement (the child wants to participate because

the activities are fun), 4) Child-directed (the child’s preferred activities are used in the session).

Therapy provides opportunities for the child to engage in sensory motor activities rich in tactile,

vestibular, and proprioceptive sensations. A key point for therapy success is the inner drive of the

child (Ayres 2005). Based on the tasks the child would like to master, the therapist designs the

environment which has to be appealing to the child and which is adapted to the child’s develop-

mental level. The treatment setting looks like a play-area with equipment designed to provide

the required form of stimulation. During these play activities the child can actively explore its

environment. The therapist assists the child to accomplish the task by manipulating the environ-

ment in such a way that the most adaptive response is elicited. Fun and success are prerequisites

for feelings of competency that are needed to achieve progress in the processing of sensory input.

During the activity the therapist observes the child’s responses and increases or decreases the

sensory and motor demands when necessary.

Over the past 30 years a number of studies have been conducted to investigate whether sensory

integration therapy is effective. The majority of studies have focused on the use of sensory inte-

gration therapy with children with learning disabilities and have aimed at improving motor skills,

academic performance, behavioural performance and/or sensory and perceptual skills. Two re-

18 19

views (Vargas and Camilli 1999, Schaaf and Miller 2005) reported that the evidence for the effects

of sensory integration is inconclusive. Bumin and Kayihan 2001 conducted a study to examine the

effects of sensory integration therapy in children with spastic diplegia. They found that children

who received the sensory integration programme performed better compared to a no treatment

control group. No studies were found which evaluated the effect of sensory integration therapy for

infants.

Functional Therapy

In the past decades the assumptions of the traditional treatment approaches have been questioned

in a considerable number of studies. Evidence for the effectiveness of the approaches is inconclu-

sive. A number of studies have shown that treatment often was not automatically transferred into

meaningful activities of daily life (Bobath and Bobath 1984, Butler and Darrah 2001, Ketelaar et al

2001). This finding initiated a conceptual shift in treatment from focusing on normality to func-

tionality. Nowadays, the concept of functional therapy is increasingly emphasized in literature on

interventions for children with CP.

The theoretical background of the functional approach is formed by the dynamic systems

theory (Thelen and Smith 1994) and family-centred service. Functional therapy consists of an

approach guided by principles of motor learning and function (Ketelaar et al 2001, Ekström-Ahl

et al 2005). Exploration and selection in finding solutions for new task demands in combination

with the adaptation to changes in the environmental context are considered to be critical parts

of motor learning (Ketelaar et al 2001). Main features of a functional approach to therapy are: 1)

Focus on the improvement of functional tasks which are problematic in daily life, 2) The child acts

as an active participant of the therapy, who gets the opportunity to actively explore his possibili-

ties and to find the best strategies and solutions for a functional task, 3) Training should take place

in a context specific environment, 4) Focus on assisting the child and his family to achieve more

satisfactory performance of daily activities (Harris 1990, Law et al 1998, Ketelaar et al 2001), 5)

Caregivers are involved in all stages of the programme (from goal-setting and implementation in

daily life to evaluation).

In the last decades the involvement of caregivers in the care process has become the basis of

the care approach in rehabilitation. This concept of family centered care (FCC) is described as a

holistic approach towards service delivery, in which the unique strengths, resources and set of

needs of each child and its family form the base for a highly individualized and dynamic model

of care (King et al 2004, MacKean et al 2005). Keywords in FCC are partnership, information

exchange and respectful and supportive treatment (King et al 1997, Rosenbaum et al 1998). Care-

givers are considered as equal partners who work together with the rehabilitation team to

optimize their child’s care process. This implies that caregivers are involved in all stages of the

rehabilitation program, from goal-setting and implementation to evaluation. However, (Law et

al 2003) state that true FCC gives caregivers the opportunity to decide their own level of involve-

ment. The intervention programme must be adapted to families’ capabilities, their situation and

daily schedule and not vice versa. This view is supported by Nijhuis and coworkers (2007) who

showed that to provide true FCC a family-specific evaluation of FCC, i.e. making an accurate

inventory of the specific needs and wishes of the individual families, is essential to avoid possible

discrepancies between the views of caregivers and health care professionals.

Research has indicated that the involvement of caregivers in the care process can be associated

with positive effects both on the development of the child (Ketelaar et al 1998, Law et al 1998, King

et al 2004) as well as on the caregivers in terms of an increase in parental skills, improved well-

being and a decreased level of parental stress (King et al 1996, Rosenbaum et al 1998, Jansen et al

2003, King et al 2004, Raina et al 2005). Caregivers can play a major role in assisting their children

to reach their maximum potential by training the transfer of functional skills form one context to

another. Moreover, the involvement of caregivers in their child’s intervention may improve their

understanding of their child’s development (Palmer et al 1988). From the literature it is assumed

that daily home-based physical therapy provided by the parents is more efficient than physical

therapy given by a therapist once a week for a half hour (Ketelaar et al 1998).

Evidence for the efficacy of functional therapy is slowly increasing. One of the first studies that

evaluated a somewhat functional approach was an often cited study by Palmer et al (1988). Forty-

eight infants, aged 12 to 19 months, were randomly assigned to receive an infant stimulation

programme including motor, sensory, language and cognitive activities of increasing complexity

or NDT. The infant stimulation programme turned out to be more beneficial for developmental

outcome than the NDT approach. Another study which showed that functional physical therapy

was beneficial for children with cerebral palsy was written by Ketelaar et al (2001). They showed

in 55 children with CP aged 2 to 7 years that the children who received the functional therapy

programme (n=28) after the intervention period improved more on functional skills in daily situ-

ations than the control group. In 2005 a pilot study was published which evaluated the effects

of a functional therapy programme on motor development and functional outcome measures of

preschool children with CP (Ekström Ahl et al 2005). Fourteen children aged 18 months to 6 years

participated in the five-month intervention. The study showed that after the programme the

children had better gross motor function and performed better on daily activities. Also in relation

to the children’s mobility a decrease was found in the amount of caregiver assistance. A major

drawback of this pilot study however was the lack of a control group. At the moment a large multi-

centred RCT is running on the effects of a functional therapy programme for young children with

CP in Canada (Law et al 2007). The study aims at an inclusion of 220 children with CP aged 12

months to 5 years.

Initially, the functional approach was developed for children with developmental disorders.

Applying this concept to young infants (less than 18 months of age) implies that therapy mainly

focuses on the encouragement of self produced motor behaviour (Resnick et al 1987, Palmer et al

1988, Ketelaar et al 2001, Ekström-Ahl et al 2005). Infants act as active participants of the therapy

(Damiano 2006, Callahan et al 2006) who get the opportunity to actively explore their motor pos-

20 21

sibilities and to find an appropriate solution for any given motor task. The therapist coaches the

caregiver to stimulate the infant to perform tasks that are both challenging and meaningful with

appropriate success and failure rates (Dirks and Hadders-Algra 2003). The focus lies on assisting

the family to achieve more satisfactory performance of daily activities (Harris 1990, Law et al

1998, Ketelaar et al 2001).

Concluding remarks

Paediatric physical therapy for infants at high risk for a developmental motor disorder can be

considered as a field in development. Based on findings that evidence for the effectiveness of the

traditional EI programmes (NDT, treatment according to Vojta, and Sensory Integration Therapy)

is limited, shifts in the application of therapy were initiated. The most important advances are

functionality instead of normality, the infant as an active participant of the therapy and the inclu-

sion of the family.

aims and outline of this thesis

The advances in paediatric physical therapy show that the physiotherapeutic guidance of infants

with or at high risk for a developmental motor disorder is very diverse. The evidence for the effec-

tiveness of the intervention programmes is inconclusive. Most studies have evaluated the effects

of early intervention at the time during infancy or preschool age when the condition is already

expressed in dysfunction. As mentioned before, the term early intervention in this thesis is limited

to paediatric physical therapy for infants up to 18 months corrected age. Therefore, the first aim of

this thesis was to review the effects of early physiotherapeutic intervention on motor and cogni-

tive development of infants with a high risk for developmental motor disorders aged less than 18

months. Two systematic reviews were performed to gain more insight in the currently available

evidence on early intervention. In these reviews specific attention is paid to unravelling elements

in intervention programmes which might contribute to a beneficial effect on motor or cognitive

development. The results of these reviews are presented in Chapter 3 and 4.

The results of both reviews indicated that intervention programmes with an onset after term

age which focus on the stimulation of self-produced motor behaviour can exert a beneficial effect

on motor and cognitive development. This knowledge served as a base for the development of a

new type of physiotherapeutic intervention, COPCA. COPCA is the abbreviation of Coping with

and Caring for infants with special needs – a family centered programme (Dirks and Hadders-Al-

gra 2003). The rationale behind the COPCA programme is presented in Chapters 2 and 6. To evalu-

ate the effects of COPCA on the development of infants at high risk for a developmental disorder

an early intervention project VIP (in Dutch “vroegtijdig interventie project”) was designed. In the

VIP project the effects of the COPCA intervention on neuromotor and cognitive development

were evaluated and compared to the effects of the traditional approach towards traditional infant

therapy (TIP). For a description of the study design, subjects and outcome measures involved in

the early intervention project VIP see Chapter 2.

The second aim of this thesis was to evaluate the effects of COPCA and TIP on developmental

outcome. The results of this evaluation are presented in Chapter 4, 7, and 8. In Chapter 4 some

preliminary results on developmental outcome for the first half of the study group are described.

Chapter 7 has a twofold focus. In the first part of the study the effects of COPCA and TIP on the

developmental outcome at 6 and 18 months are described. In the second part the findings of the

randomized controlled trial were extended with the findings on the contents of the intervention

(described in Chapter 5 and 6). In this way we attempted to identify whether changes in develop-

mental outcome were associated with the contents of the intervention or that they could be ex-

plained by other factors. Chapter 8 discusses the outcomes of the VIP project including some data

which have not been published earlier.

The third aim of this thesis was to evaluate the application of COPCA in daily practice by

means of video analysis. This process evaluation is a prerequisite for the implementation of

COPCA within paediatric physiotherapy. Through detailed analysis of videos of physiotherapeu-

tic intervention sessions we gathered knowledge on the actual contents of both the COPCA and

TIP intervention. This analysis will help to discover the effective and ineffective elements in the

interventions and will consecutively result in knowledge about the implementation of intervention

by different paediatric physical therapists in different settings. Chapter 5 introduces an observation

protocol which we developed for the analysis of actions that physiotherapists use during treat-

ment. Eight main categories of physiotherapeutic actions were defined to explore the contents

of COPCA and TIP. Chapter 6 reports the contents of TIP and COPCA. Here the similarities and

differences between COPCA and TIP are lined out.

The thesis concludes with a general discussion of the reported findings focusing on implica-

tions for further research and practice.

22 23

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C H a P t E r 2

t h e

‘ V roe g t I j dIg

I n t e rV e n t I e

P roj e c t ’ ( V I P)

26 27

Dirks and Hadders-Algra (2003) developed a new physiotherapeutic intervention programme,

named COPCA, for infants at high risk for a developmental motor disorder. COPCA stands for

COPing with and CAring for infants with special needs – a family centred programme. The

COPCA intervention is based on new insights in the field of education and family care and in

the normal and abnormal development of the central nervous system. The effects of COPCA on

the development of infants at high risk for a developmental motor disorder were evaluated in the

early intervention project VIP (in Dutch “vroegtijdig interventie project”). The VIP project was

started in 2003 and the results are presented in this thesis (see Chapter 4,5,6,7, and 8). Infants who

had been admitted to the Neonatal Intensive Care Unit (NICU) of the University Medical Center

Groningen between March 2003 and May 2005 and showed definitely abnormal General Move-

ments (GM) were eligible for this study. The presence of definitely abnormal GMs indicates a high

risk for developmental motor disabilities, such as cerebral palsy (CP) (Prechtl 2001, Hadders-Algra

2004). Infants with severe congenital anomalies and infants whose parents had an inappropriate

understanding of the Dutch language were excluded from the study. The VIP project was designed

as a randomized controlled trial. Through block randomization infants were assigned to the ex-

perimental group who received the COPCA intervention or to the control group who got the tradi-

tional form of infant physical therapy (TIP). The second goal of the VIP project was to evaluate

the application of COPCA in daily practice by means of video analysis. This process evaluation is a

prerequisite for the implementation of COPCA within paediatric physiotherapy at a later stage.

In the next section a brief outline of the TIP and COPCA intervention is provided. The

remaining sections of this chapter describe the study design, subjects and outcome measures

involved in the early intervention project VIP. The outcomes of the VIP project are described in

Chapter 4, 5, 6, 7, and 8.

tIP

TIP consists of infant physical therapy as it is nowadays applied in the Netherlands. TIP includes

treatment strategies from a variety of physiotherapy programmes such as NDT, treatment accord-

ing to Vojta and the Functional approach. For a description of these programmes see Chapter 1.

For the most part TIP consists of the implementation of the ‘living concept’ of neurodevelopmen-

tal treatment (NDT; Howle 2002). Influences from the functional approach, such as the encour-

agement of self produced motor behaviour, are more and more incorporated into the treatment.

Due to the different influences which are incorporated in TIP a large heterogeneity in treatment

application can be expected.

COPCa

COPCA is a relationship family focused intervention programme built on two theoretical com-

ponents. The first component is based on the transactional model of development (Sameroff and

Chandler 1975, Fiese and Sameroff 1989) and recent findings in the field of education and family

care (Dale 1996, Law et al 1998, Rosenbaum et al 1998). The second component, a neurodevelop-

mental one, is based on the principles of the Neuronal Group Selection Theory (NGST; Edelman

1993).

An important base of COPCA is formed by the transactional model of development (Sameroff

and Chandler 1975, Fiese and Sameroff 1989) and new insights in the field of education and family

care (Dale 1996, Law et al 1998, Rosenbaum et al 1998). In the transactional model development

is seen as a result of a continuous dynamic interplay among child behaviour, caregiver responses

to the child’s behaviour, and environmental variables that may influence both the child and the

caregiver. Therefore in COPCA optimizing the interaction between caregiver and child is a key

factor of the intervention. The physiotherapist can be seen as an environmental factor that may

influence both the child and the caregiver. The physiotherapist acts as a coach who respects the

autonomy of the family and intends to make him- or herself superfluous. Caregivers are key-

persons in the COPCA programme. The physiotherapist and the family are partners, in which the

family defines the priorities for intervention, while the therapist coaches the family throughout

the intervention period (Law et al 1998). The PT provides information on infant development, the

specific problems of infants with neurological dysfunction, basic principles on infant education.

Specific ways in which development may be promoted and problems may be tackled. By means of a

continuous dialogue with the PT, caregivers develop their own ways to cope with the problems of

the infant.

The second component of COPCA is based on the principles of the NGST. The NGST em-

phasizes that development is the consequence of a complex interaction between genetic infor-

mation and environmental influences. According to the NGST development is characterized by

two phases of variability, i.e. primary and secondary variability. During primary variability the

child explores all variations of motor possibilities that are available in the nervous system. In this

phase the child is not yet able to adjust his behaviour to external conditions. In typically develop-

ing infants this phase is characterized by abundant variation. At function specific ages the infant

reaches the phase of secondary variability. The child gradually learns to select the most efficient

solution for a given task out of his motor repertoire. This selection is based on trial and error

experiences (Hadders-Algra 2000a). This means that during the phase of secondary variability

the child learns to adapt his/her motor behaviour to specific situations. Infants with a pre- or

perinatally acquired brain lesion resulting in a developmental motor disorder such as CP have a re-

duced repertoire of motor strategies available. This is already expressed during the first postnatal

months in a limited repertoire of general movements and continues when goal-directed motility

emerges (Hadders-Algra 2000b). In addition, these infants have problems with the selection of the

most appropriate solution for a certain task out of the repertoire due to deficits in the processing

of sensory information (Hadders-Algra 2000b). Therefore COPCA aims to promote variation in

motor behaviour and trial-and-error experiences by the means of play. The ultimate goal is to

provide an infant with a larger diversity in neuronal networks that will help to find an appropriate

solution for any given motor task.

28 29

Subjects and Procedure

The study groups of the VIP project consisted of infants who had been admitted to the Neonatal

Intensive Care Unit (NICU) of the University Medical Center Groningen between March 2003

and May 2005. At ten weeks corrected age (CA) a video recording was made of the infants’ General

Movements (GM). The assessment of the quality of GM can be considered as a technique to evalu-

ate the quality of the brain function in young infants (Prechtl 1990, 2001, Hadders-Algra 2004).

General Movements

GM are spontaneously generated complex movements involving the head, trunk, arms and legs.

They emerge at seven to eight weeks postmenstrual age and remain the most frequently observed

movement pattern until the age of three to four months after term age when they gradually are

replaced by goal-directed motor behaviour (Hadders-Algra 1996, 2001). Four classes can be distin-

guished in the assessment of GM quality: normal-optimal, normal-suboptimal, mildly abnormal

and definitely abnormal GM. In typical development GM are characterized by a considerable vari-

ation and complexity and to a smaller extent by fluency. When GM are less optimal fluency is the

first aspect that disappears, the movements become more jerky or stiff. Variation and complexity

disappear when the movements become more abnormal. Various studies have shown that the

presence of definitely abnormal GM at the age of two to four months corrected age (CA), the so-

called fidgety GM phase, is associated with a high risk for developmental motor disorders such as

CP (Prechtl et al 1997, Prechtl 2001, Hadders-Algra 2004, Groen et al 2005). Research showed that

the GM method has a good predictive validity for both major and minor developmental disorders

Heineman and Hadders-Algra, 2008). Hadders-Algra (2004) showed that the infants who show

definitely abnormal GM, but do not develop a CP usually show other developmental problems,

such as minor neurological dysfunction (MND) and behavioural problems. Also the construct and

concurrent validity and reliability of the GM method are satisfactory (Heineman and Hadders-

Algra 2008). The early detection of infants at high risk for developmental disorders offers the

opportunity for intervention at young age, during a phase in which the central nervous system is

characterized by high plasticity (Kolb et al 2001, De Graaf-Peters and Hadders-Algra 2006). Plas-

ticity means that the brain can reorganize itself to some extent. Neurons neighbouring a lesioned

part can change function and become incorporated into the affected repertoires, thereby increas-

ing the variation of the movement repertoire of the child (Hadders-Algra 2000b).

During the period of March 2003 till May 2005 in total 257 video recordings of GM were made

for the VIP project. Infants and their families were selected to participate in the project when

the quality of the infants’ GM was classified as definitely abnormal. Sixty-two infants showed

definitely abnormal GM (Figure 2.1). Infants with severe congenital anomalies and infants whose

caregivers had an inappropriate understanding of the Dutch language were excluded from the

study. Forty-six families decided to participate in the VIP project. Through block randomization

(full term infants blocks of n=2; preterm infants blocks of n=12) the infants were assigned to the

experimental group who received the COPCA intervention (n=21) or to the control group who got

the traditional form of paediatric physical therapy (TIP; n=25). The randomized intervention was

provided between 3 to 6 months corrected age (CA). The COPCA intervention was carried out by

4 physiotherapists, who were intensively trained in the application of this method. COPCA was

applied twice a week for one hour in a home based setting. The application of TIP varied. Infants

received therapy with a median value of once a week, the mean duration of sessions was 30 min-

utes. TIP was also mainly applied in the home situation by the paediatric physiotherapist working

in the area. After the intervention period physical therapy was only continued when the paedia-

trician in charge of the care of the infant considered it necessary. In the TIP group were three

infants who did not receive physiotherapeutic guidance during the intervention period since their

paediatrician did not refer them to a physiotherapist.

Figure 2.1. Infants included into the VIP project.

Total number of infant GMs March 2003 – May 2005N = 257

Definitely abnormal GM qualityN = 62

RandomizedN = 46

COPCAN = 21

Assessed at 18 mo CAN = 21

TPPN = 25

Assesses at 18 mo CAN = 232 did not attend

Not randomizedN = 161 non-Dutch speaking parents3 severe congenital anomalies12 parental refusals

30 31

Evaluation and Outcome measures

The developmental status of the infants was assessed at 3, 4, 5, 6 and 18 months CA. Table 2.1

shows which outcome measures were assessed on the different evaluation moments. Neurologi-

cal condition at 3 to 6 months was measured with the Touwen Infant Neurological Examination

(TINE; Hadders-Algra et al 2009). At 18 months CA we used the neurological examination accord-

ing to Hempel (1993). Additionally, at 18 months CA the concept of neurological optimality was ap-

plied to detect subtle differences in neurological condition (Huisman et al 1995). The Infant Motor

Profile (IMP; Heineman et al 2008) was used to assess the neuromotor condition in a qualitative

way. This instrument pays attention to the evaluation of motor abilities, movement variability,

the ability to select motor strategies, movement symmetry, and fluency. Gross motor development

was evaluated with the Alberta Infant Motor Scale (Piper and Darrah 1994). For the cognitive

development of the infants the mental scale of the Bayley Scales of Infant Development was used

(Bayley 1993, Van der Meulen et al 2002). During the assessments at 4, 6 and 18 months CA we also

gathered information about the development of postural control of the infants.

Furthermore, we administered questionnaires to investigate the psychosocial development of

the infants; the Kent Infant Development Scale (KIDS) (Schneider et al 1990, Reuter and Gruber

2000) and the Pediatric Evaluation of Disability Inventory (PEDI) (Haley et al 1992, Custers et al

2002a). Information about education of the infants was obtained with the Nijmegen Child-rearing

3 months 4 months 5 months 6 months 18 months

TINE TINE TINE TINE Hempel + NOSIMP IMP IMP IMP IMPAIMS AIMS AIMS AIMS AIMS

BSID –II BSID- IIPostural control Postural control Postural control

KIDS KIDS KIDSPEDI PEDI PEDINCSQ NCSQ NCSQ

MPOC MPOC MPOCVideo PT Video PT

Video ADL Video ADL Video ADL

table 2.1. Outcome measures.

AIMS = Alberta Infant Motor Scale, BSID-II = Bayley Scales of Infant Development, 2nd edition, IMP = Infant Motor Profile, KIDS = Kent Infant Development Scale, MPOC = Measures of Processes of Care, NOS = neurological optimality score, NCSQ = Nijmegen Questionnaire on Child-rearing Situations, PEDI = Pediatric Evaluation of Disability Inventory, TINE = Touwen Infant Neurological Examination, Video ADL = video of activities of daily living, Video PT = video of physical therapy session.

Situation Questionnaire (NCSQ) (Wels and Robbroeckx 1996). The extent to which caregivers

perceive the care they received as family centred and the contentment of the caregivers with the

physiotherapeutic care was measured with the Measures of Processes of Care (MPOC; King et al

1995, van Schie et al 2004). The contents of the intervention and the application in daily life were

evaluated by the use of video-analysis. Therefore we made videorecordings of intervention ses-

sions at 4 and 6 months CA and recordings of activities of daily living (ADL) at 3, 6 and 18 months

CA. A short description of the outcome measures is provided in this paragraph.

Neurological examination

To evaluate the neurological condition of the infants we used the Touwen Infant Neurological

Examination (TINE; Hadders-Algra et al 2009). The neurological findings were summarized as

normal, normal-suboptimal, minor neurological dysfunction (MND) or abnormal. During infancy

the classification of abnormal implies the presence of a distinct neurological syndrome, such as

a clear hypo- or hypertonia, a hemisyndrome, or a hyperexcitability syndrome. To distinguish

between MND, normal-suboptimal and normal neurological condition, the findings of the TINE

are classified according to age-specific norms into five clusters of dysfunction: dysfunctional

reaching and grasping, dysfunctional gross motor function, brain stem dysfunction, visuomotor

dysfunction and sensorimotor dysfunction. Two forms of normal neurological development can

be distinguished: neurologically normal when none of the clusters meets the criteria for dysfunc-

tion and normal-suboptimal when one or two clusters fulfil the criteria for dysfunction. When

more than two clusters fulfil the criteria for dysfunction infants are classified as MND. A recent

study showed that MND can be assessed reliably with the TINE (Hadders-Algra et al 2009).

Neurological examination according to Hempel and Neurological Optimality Score (NOS)

The Hempel assessment is a standardized assessment technique designed for the detection of

signs of minor neurological dysfunction during toddler age (1.5-4 years) (Hempel 1993). But of

course, the assessment also allows for the evaluation of pathological conditions. Findings of the

Hempel assessment were classified as neurologically normal, simple MND, complex MND or

neurologically abnormal indicating conditions such as CP. The distinction between simple and

complex MND is also based on the number of clusters of dysfunction. Simple MND at 18 months

denotes the presence of one cluster of dysfunction and complex MND the presence of more than

one cluster of dysfunction. The classification CP implies the presence of a ‘classical’ configuration

of neurological signs (Hadders-Algra 2003).

Not only the traditional signs of neurological dysfunction, for example mild abnormalities in

muscle tone or reflexes and motor milestones are assessed with the Hempel assessment. Through

observation of spontaneous motor behaviour in a free field play situation the investigator can

observe the qualitative aspects of spontaneous motor behaviour without compromising the

cooperation of the child (Hempel 1993, Hadders-Algra 2005). The Hempel assessment consists of

94 items covering 7 sections including prehension function, sitting behaviour, crawling behaviour,

32 33

standing and walking behaviour, evaluation of cranial nerve functions, and sensorimotor

function by means of manipulation including the assessment of muscle tone and the intensity and

threshold of reflexes. Little information is available about the concurrent and predictive validity of

the Hempel assessment. The interrater reliability is satisfactory (Hempel 1993).

Additionally at 18 months CA we used the Neurological Optimality Score (NOS) (Huisman et

al 1995) to summarize neurological condition. Based on the neurological examination an optimal-

ity score can be calculated to detect small deviations in neurological condition. The NOS is the

sum of 57 items representing the neurological examination that meet predefined criteria for opti-

mality. Note that the definition of optimal is narrower than that of normal or typical, and that a

reduced optimality not always means abnormal (Prechtl 1980). The NOS has been proven to be an

excellent instrument to evaluate subtle differences in neurodevelopmental outcome (Bouwstra et

al 2006).

Infant Motor Profile

The IMP is a video-based assessment of motor behaviour in infancy (Heineman et al 2008). The

IMP consists of 80 items that evaluate motor abilities, movement variability, the ability to select

motor strategies, movement symmetry, and fluency. It is applicable in children from 3 to 18

months. The advantage of the IMP is that it evaluates motor behaviour in a qualitative way. The

IMP has sound psychometric properties. The intra- and interobserver agreement are satisfactory

and the concurrent validity with the AIMS was good. Moreover the IMP was able to differentiate

between infants with normal neurological condition, simple MND, complex MND, and abnormal

neurological condition (Heineman et al 2008). No information is available on the predictive vali-

dity of the IMP.

Alberta Infant Motor Scale

The Alberta Infant Motor Scale (AIMS; Piper and Darrah 1994) is an observational instrument to

measure gross motor development for infants from birth to independent walking. The AIMS con-

sists of 58 items and is organized into four positions: prone, supine, sitting and standing. Through

observation of spontaneous motor behaviour each item can be scored on three aspects of motor

performance, namely weight bearing, posture and antigravity movements. The AIMS can be used

for two purposes. First, to identify infants who are delayed or atypical in their motor performance

and second to evaluate motor development over time (Piper and Darrah 1994). The AIMS has very

good reliability coefficients (Piper and Darrah 1994, Heineman and Hadders-Algra 2008). Also the

validity of the AIMS is well examined and proven to be satisfactory (Piper and Darrah 1994, Dar-

rah et al 1998, Liao and Campbell 2004).

Bayley Scales of Infant Development

The Bayley Scales of Infant Development (BSID-II; Bayley 1993) is a psychometric sound instru-

ment to assess mental and psychomotor development of children aged 1 to 42 months. In the early

intervention project the Dutch version of the BSID-II was used to assess the cognitive develop-

ment at the age of 6 and 18 months CA (Van der Meulen et al 2002). The BSID-II consists of a

mental developmental index (MDI) and a psychomotor developmental index (PDI). A limitation of

the BSID-II psychomotor scale is that the emphasis is placed on cognitive motor performances.

Therefore we decided to administer only the MDI for the VIP project and to assess gross motor

development with the AIMS and the qualitative aspects of motor performance with the IMP. The

MDI was scored based on the items successfully completed. The MDI consists of items concern-

ing problem solving, memory, discrimination, classification, language and social skills. Since the

children were not exactly 6 or 18 months at time of the assessment, the scores were converted

into age-equivalent scores, as derived from the Dutch norms (van der Meulen et al 2002). The

interrater reliability of the MDI is sufficient, the construct and concurrent validity are moderate

(Heineman and Hadders-Algra 2008).

Postural control

The development of postural control was assessed, because infants with high risk for developmen-

tal disorders often have problems with their postural control which interfere with the activities

of daily life (Brogren et al 2001, Van der Heide et al 2004). Postural activity was assessed at 4 and 6

months CA during reaching while lying in supine and while sitting with support, and at 18 months

CA while sitting with support and – when possible – without support. Reaching was elicited by

presenting toys when the infant was in a calm and alert behavioural state. Elecromyografic (EMG)

recordings were measured continuously during the testing session with bipolar surface electrodes

placed on the following muscles: deltoid, pectoralis major, biceps brachii, triceps brachii, neck

flexor (m. sternocleidomastoid), neck extensor, rectus abdominis, thoracal extensor, lumbar ex-

tensor, rectus femoris, and hamstrings on the right side of the body. Simultaneous with the EMG

recordings, movements were recorded kinematically. For a detailed description of the procedure

see de Graaf-Peters et al (2007).

Kent Inventory of Developmental Skills (KIDS)

The Kent Inventory of Developmental Skills is a caregiver-completed developmental assessment

instrument for healthy infants, infants at risk, and for young children with developmental disabil-

ities who are chronologically or developmentally under the age of 15 months (Schneider et al 1990,

Reuter and Gruber 2000). The KIDS was used as comparison to the BSID-II. Both measures aim to

evaluate the developmental level of the infant. At 3 months CA only the KIDS was administered to

minimize the physical effort of the infant. At 6 and 18 months CA both instruments were assessed,

being aware that the KIDS at 18 months might suffer from a ceiling effect. Parents were asked to fill

out the questionnaire at home at 3,6, and 18 months CA and return it to the researchers.

The main idea of the KIDS is that all new behaviours are acquired in a more or less fixed order

which is age dependent. The KIDS comprises of 252 behavioural items which are divided into

five areas: motor behaviour (78 items), cognition (52 items), social behaviour (51 items), ability to

34 35

perform things independently (39 items) and language (38 items). Caregivers fill in whether or not

their child shows the described behaviour. The total score provides an impression whether the

developmental status of the child corresponds with the average chronological age of its peers with

similar behavioural repertoires, and the status of the child as having no delay, at risk or develop-

mentally delayed. Research showed that the interrater agreement, test-retest reliability and vali-

dity were high (Schneider et al 1990, Reuter and Gruber 2000). As the KIDS is a parent reported

questionnaire it can be discussed whether caregivers are capable to objectively judge the develop-

ment of their infant. Research has shown that most caregivers are very well capable of judging

their infants’ developmental level (Harris 1994, Johnson et al 2008). Therefore we assume that this

is also the case in the VIP project.

Pediatric Evaluation of Disability Inventory

The Pediatric Evaluation of Disability Inventory (PEDI; Haley et al 1992) was used to measure the

functional ability of the child. This assessment instrument is developed for young children from

6 months to 7.5 years of age with developmental disabilities and adapted to a Dutch version by

Custers et al (2002a). The PEDI is a discriminative measure which aims to detect whether a child

has a delay in his functional status and if so to determine the extent and content area of the delay.

With the PEDI both the capability of the child (what a child can do) and the performance (what

the child actually does do) of routine daily activities can be evaluated. Capability is measured by

the functional skills scale (197 items), while the caregiver assistance scale and the modifications

scale provide information on the performance (20 items). Each scale consists of three domains

namely self-care, mobility and social function. The PEDI can be considered as a reliable and valid

instrument (Custers et al 2002a, 2002b, Wassenberg-Severijnen et al 2003).

Measures of Processes of Care (MPOC)

The MPOC is designed to determine the extent to which caregivers perceive the health services

they receive as family centred (King et al 1995, 1997). In the VIP project we used the Dutch trans-

lation of the MPOC (Van Schie et al 2004). Additionally, the MPOC was also used to evaluate

whether the attitude of caregivers regarding health services changed over time. The MPOC is a

56-item self-administered questionnaire categorised in five domains: a) enabling and partnership,

b) providing general information, c) providing specific information about the child, d) coordinated

and comprehensive care, e) respectful and supportive care. The items ask to what extent a particu-

lar behaviour occurred during the past year. The responses are given on a 7-point scale ranging from

‘not at all’ to ‘to a very great extent’ with an additional ‘not applicable’ category. Domain scores are

calculated as the mean of the ratings for the items in the scale and is only considered as valid when

at least two third of the items in the scale received a score. The validity criterion for the question-

naire was completion of at least half of the items. The Dutch translation of the MPOC has sound

psychometric properties; the construct validity and reliability measures are good (Van Schie et al

2004).

Nijmegen Child-rearing Situation Questionnaire (NCSQ)

The Nijmegen Child-rearing Situation Questionnaire (NCSQ, Wels & Robbroeckx 1996) was used

to investigate the existing parenting situation. The questionnaire is based on the stress model

of Lazarus and the attribution theory of Weiner (Wels and Robbroeckx 1996). The instrument

consists of 4 scales: A) Subjective Family Stress. This scale consists of 46 items which form eight

categories: a) acceptance of the child, b) coping, c) experience problems, d) need for change, e)

child is a strain, f) managing on one’s own, g) having pleasure, h) relation. B) Assessment of the

childrearing situation. Parents are instructed to indicate which of eight descriptions fits best

their own situation. In the first situation, no problems in child-rearing are present and support

is absolutely unnecessary. In situation eight, child-rearing is highly problematic and parents are

in urgent need of support. C) Attributions on the Child-rearing situation. This scale consists of

34 items which form five categories: a) ability, b) effort of oneself and partner, c) task difficulty, d)

share of partner, e) luck/fate. D) Expectation for help. This last scale has 36 items divided over five

categories: a) satisfaction, b) need for change, c) need for help, d) expectation for help (internal

orientation), and e) expectation for help (external orientation). The answers of the y questions in

scale A, C and D are rated on a 5-point Likert scale, ranging from 1 = ‘‘does not apply to me at all’’

to 5 = ‘‘applies to me’’. Sum scores were calculated for each category and converted into weighted

scores. Higher scores indicate that the caregivers experience a relatively greater burden on the

child’s education. The reliability and the validity are sufficiently assessed (Wels and Robbroeckx

1996). Note however that later research showed that the norms are insufficiently assessed, because

the reference groups were too small (Evers et al 2000). The caregivers are asked to fill in the NCSQ

at 3, 6 and 18 months corrected age.

Video recordings of physical therapy sessions and ADL activities

During the VIP project video recordings were made of physiotherapeutic intervention sessions

and of daily caregiving tasks. Albrecht et al (2005) showed that video recording hardly affects the

behaviour of people that are being filmed, because they tend to forget the camera after a while. In

order to ensure that the recording would resemble a natural situation and to minimize intrusion

we took the following precautions: the camera was positioned as far away from the physiothera-

pist, caregiver and infant as possible and the physiotherapist and caregiver were asked to ignore

the person making the video.

Video recordings of intervention sessions were made at 4 and 6 months CA. They were used to

gain knowledge on the contents of paediatric physiotherapeutic intervention. On the one hand,

this clearly showed whether the contents of COPCA were significantly different from the contents

of TIP. On the other hand, the analysis of the video recordings enabled us to answer the question

whether the COPCA programme could be well implemented in daily physiotherapeutic practice

(see for more detail Chapter 5 and 6).

The video recordings of activities of daily living – i.e. bathing and playing – showed how well

principles of the COPCA programme were implemented in the daily life of the families. These

36 37

activities were recorded at 3, 6 and 18 months CA. In this thesis the analysis and results of the ADL

video recordings will not be further described.

The outcomes of the VIP project are presented in Chapter 4, 5, 6, 7, and 8. In Chapter 4 some pre-

liminary results of the first half of the study group on the neurological examination, NOS, AIMS

and MDI of the BSID-II are described. Chapter 5 and 6 report the results of the video analysis on

the contents of paediatric physiotherapeutic intervention, i.e. COPCA and TIP. In Chapter 7 the

developmental outcome of the infants on the neurological examination, NOS, AIMS, MDI of the

BSID-II and PEDI is described. Subsequently, to examine whether developmental outcome could

be associated with treatment principles used by the PT or caregiver during intervention or with

confounding factors we applied process evaluation to the data. Finally, in Chapter 8 an overview

of the results on outcome measures which were also assessed during the VIP project, but were not

the focus in the previous Chapters is provided. Attention is paid to the results of the following

questionnaires: KIDS, MPOC, and NQCS. Also some preliminary results on the postural control

of the infants are described. Data of the IMP, the ADL video recordings and postural control are

currently being assessed.

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C H a P t E r 3

A s ys t e m At Ic r e V I e w

of t h e e f f e c t s of

e A r ly I n t e rV e n t Ion on

mo tor de V e l oPm e n t--------------------------------------------------------------------------------------------

CH BLAUW-HOSPERS, M HADDERS-ALGRA

Developmental Medicine and Child Neurology, 2005, 47; 421-432

40 41

abstract

We present a systematic review on the effect of early intervention starting between birth and the

corrected age of 18 months on motor development in infants at high risk for or with developmen-

tal motor disorders. Thirty-four studies fulfilled the selection-criteria.

Seventeen studies were applied within the NICU environment. Eight had a high methodologi-

cal quality. They evaluated various forms of intervention. The results indicated that NIDCAP

intervention might have a temporary positive effect on motor development. Twelve of the 17 post-

NICU studies had a high methodological quality. They addressed the effect of NDT and specific

or general developmental programmes. The results showed that intervention according to the

principles of NDT does not have a beneficial effect on motor development. They also indicated

that specific or general developmental programmes can have a positive effect on motor outcome.

We concluded that the type of intervention which might be beneficial for infants at preterm

age differs from the type which is effective in infants who have reached at least term age. At pre-

term age infants seem to benefit most from intervention which aims at mimicking the intra-

uterine environment, such as NIDCAP intervention. After term age, intervention by means of

specific or general developmental programmes has a positive effect on motor development.

Keywords

Early intervention, motor development, NDT, NIDCAP, infant stimulation

Introduction

In the last decades the importance of Early Intervention (EI) is generally recognized. But what ex-

actly is early intervention? Typically a single definition is used, which is applied for EI to children

at biological risk for developmental disorders and children with developmental disabilities. “Early

Intervention consists of multidisciplinary services provided to children from birth to 5 years of

age to promote child health and well-being, enhance emerging competencies, minimize devel-

opmental delays, remediate existing or emerging disabilities, prevent functional deterioration

and promote adaptive parenting and overall family functioning. These goals are accomplished

by individualized developmental, educational and therapeutic services for children in conjunc-

tion with mutually planned support for their families” (Shonkoff and Meisels 2000). In general,

EI programmes use techniques derived from the domains of physiotherapy, occupational therapy,

developmental psychology and education. Little attention is paid to the effect of nutrition, even

though it is well known that cognitive outcome of breastfed children is significantly better than

that of formula-fed children (Anderson et al 1999). The earliest studies on EI programmes prima-

rily addressed the improvement in motor skills. Later, the focus shifted towards family-focused

and other functional outcomes (Harris 1997). Thus, it appears that EI serves as an umbrella term

covering the whole field of childhood intervention.

One of the problems associated with the use of the term EI is the interpretation of ‘early’.

‘Early’ can be understood in two ways, namely as ‘early in life’ and as ‘early in the expression of the

condition’. Each of the two types of ‘earliness’ is associated with advantages and disadvantages for

intervention. The major advantage of intervening early in life is that the brain is considered to be

very plastic. The brain is especially plastic in the phase occurring after the completion of neuronal

migration during which the processes of dendritic outgrowth and synapse formation are highly

active (Kolb et al 2001). This means that high plasticity can be expected between 2 to 3 months

before until about 6 to 8 months after term age (Hadders-Algra 2001). However, there are two

potential disadvantages which might be associated with intervention early in life. First, the type of

problems the infant will develop later in life will not yet be clear. This makes effective goal setting

for EI difficult. Second, a part of the at-risk population will not develop a developmental disorder,

thereby making intervention perhaps superfluous for these children.

Intervention in children with a developmental disorder in general starts later in life, i.e. at the

time during infancy or preschool age when the condition is expressed in dysfunction. Two advan-

tages of the latter situation are that intervention is applied to children who are really in need of

EI and that the goals of the intervention can be formulated relatively easily. The most important

disadvantage of intervention which first starts when the disorder has become undeniable is that

it starts relatively late from a point of view of the plasticity of the brain (Hadders-Algra 2001).

Indeed, previous studies indicated that programmes which started before the ninth month post

term provided more improvement in both motor and personal skills than programmes starting

later (Shonkoff and Hauser-Cram 1987, Sharkey et al 1990). Thus, it seems wise to start early in

life.

42 43

Therefore, the aim of the present study is to systematically review studies on intervention starting

early in life in children at high biological risk for developmental disorders. In particular we aim

at unravelling the elements, which might contribute to a beneficial effect on motor development.

Specific attention is paid to the timing of intervention in order to see whether we can find indica-

tions for the existence of a crucial age period in which EI results in the most beneficial outcome.

Method

Selection procedure

A literature search was performed using the following electronic databases: MEDLINE (1966 to

July 2004), CINAHL (1982 to July 2004), AMED (1985 to July 2004), PsycINFO (1967 to July 2004)

and PEDro. Reference lists in original studies and reviews were also examined for appropriate

articles. The keyword ‘early intervention’ initially revealed 13,699 hits. In order to reduce the

number of hits the following additional keywords were used: ‘infant’, ‘motor development’, ‘low

birth weight’, ‘preterm’, ‘high-risk’ and ‘cerebral palsy’. Thereafter the number of hits was limited

to 485.

From these 485 papers only studies were included in the review when they fulfilled the follow-

ing four criteria: a) subjects of study: infants with high biological risk for or with developmental

disabilities, b) aim of the intervention (mostly amongst others): to improve motor development, c)

onset of intervention in at least 50% of the participating children: between birth and the (cor-

rected) age of 18 months, d) journal in which the study was published had an impact factor of > 0.3.

Excluded from the review were studies restricted to medical and orthopaedic interventions and

studies in populations of healthy low-risk preterm infants or in populations of socially disadvan-

taged children without specific biological risk for developmental disorders.

Based on the abstract, 60 papers were selected as potential candidates for the review. Reading

the entire paper left 36 original studies which met all inclusion criteria. There were two studies

within this selection of which the results were published in more than one paper (Resnick et al

1987, 1988, Palmer et al 1988, 1990). In the present review these studies were treated as single

entities. The papers not included in the review mainly were review articles, studies focusing on

improvement of cognitive function and studies in which the intervention was applied after the age

of 18 months.

Evaluation procedure

The evaluation of the studies focused on the type and size of groups included in the study, the

level of evidence (Table 3.1) and the internal and external validity of the study (criteria specified in

Table 3.2). The level of evidence of a study and its internal and external validity were the determi-

nants for the methodological quality of the study. All studies were rated independently by both

reviewers. Interrater agreement was calculated for each of the determinants of methodological

quality. Agreement was high: Cohen’s Kappa varied from K = 0.86 (internal validity), K = 0.94

(level of evidence) to K = 0.95 (external validity). Disagreements were discussed till consensus was

reached. In addition, attention was paid to specifics of the intervention programme, such as the

type of intervention, its period of application and intensity, the location where the intervention

had been carried out and parental involvement (Table 3.2). Finally, the age of the infants at evalu-

ation, the outcome measures and the results were specified to further evaluate the effect of the

intervention.

In order to assess the effect of the age period during which EI took place, we divided the stud-

ies into three age-groups (cf. Shonkoff & Hauser-Cram 1987, Sharkey et al 1990). The first group

consisted of a) studies dealing with intervention programmes applied in the Neonatal Intensive

Care Unit (NICU), b) the second group contained studies where the intervention programme

started between discharge from NICU care and 9 months corrected age, c) the third group in-

cluded studies where intervention started between 9 and 18 months corrected age.

The studies turned out to be so heterogeneous in type of intervention evaluated, outcome

measures used to evaluate the effect of intervention and age at which outcome was studied, that

no meta-analysis to examine effect sizes of interventions could be performed.

table 3.1. Levels of evidence (Sackett 1989, Butler and Darrah 2001).

Level Group research Single individual research

I Randomised controlled trials N=1 randomised controlled trialII Non-randomised controlled trials

Prospective cohort studies with concurrent control groupABABA designAlternating treatments (e.g. ABACA)Multiple baseline across participants

III Case studies with control participantsCohort studies with historical control group

ABA design

IV Case series without control participants AB designV Case reports

Non-empirical methodsCase reportsNon-empirical methods

results

Seventeen of the 34 studies dealt with interventions carried out within a NICU setting, eight

evaluated intervention starting between discharge from the NICU and 9 months and six studies

assessed intervention which started between 9 and 18 months corrected age. In three studies, the

age at onset of intervention exceeded the pre-set period criteria. Therefore, a fourth category of

age at onset of intervention was added, consisting of studies where intervention started at some

age between discharge from the NICU and 18 months corrected age. Within the age periods, stud-

ies were ranked primarily according to level of evidence, next based on internal validity, external

validity and effect (Tables 3.3-3.6).

44 45

Group & design DescriptionSample size

?Total, Experimental group, control or contrast groupNot present

Attrition?

NumberUnspecified

Study group?

High-risk (i.e. preterm/LBW), CP, DownUnspecified

Levels of evidence Levels according to Sackett’s method for grading research (Sackett, 1989) modified by Butler and Darrah (2001); see Table 3.1.

Internal validity: can measured effects be attributed to intervention under study?

+++-

High internal validityFair internal validityLow internal validityInternal validity can be reduced by various study variations such as subject assignment, contamination, co-intervention, and blind assessment

External validity: can results of research be generalized?

+++-

Generalization is plausibleSome possibilities for generalizationNo information about generalization

Methodological quality Based on level of evidence, internal and external validity

Intervention Method of treatmentContents description ++

+-

Detailed descriptionSummary onlyVery limited information

Period of application?

Description Unspecified

Intensity?

DescriptionUnspecified

Location Hospital, Home, CentreCombination

Parental involvement PT?

Enhance parental skillsParent is therapistUnspecified

Evaluation of effectAge at evaluation < term, around term, 1-9 mo post term, 9-18 mo, > 18 mo Outcome measures Neuromotor

DevelopmentalResults E=C, E>C, E<C

table 3.2. Evaluation criteria (modification of Siebes and Vermeer 2002). Populations and methods of intervention

The number of children included in the studies ranged from 10 to 746 individuals (median value:

n= 44.5; Table 3.3). Information about the rate of attrition was provided in 33 studies. It varied

from 0% to 64%, with a median value of 13%. In the majority of studies (24 out of 34) the study

group consisted of so-called high-risk infants, i.e. infants born preterm or infants with a low birth

weight. The remaining studies evaluated the effect of intervention in infants with cerebral palsy,

delayed mental and motor development or Down syndrome.

The intervention programmes applied showed considerable diversity (Table 3.4). In 29 studies,

at least brief information about the intervention method was given of which 10 studies gave a

more detailed description. In order to get some insight into the intervention strategies and pro-

cedures applied, we assessed whether programmes contained the following elements: procedures

to reduce stress, sensory stimulation (specific unimodal, specific multimodal, general multimo-

dal), motor intervention strategies (passive handling techniques, active training of specific motor

abilities, general motor training) and parent infant interaction strategies (Table 3.6). Both authors

assessed the composition of the intervention programmes independently. Interrater agreement

was high: Cohen’s Kappa for the various components varied from 0.79 to 1.00. NICU intervention

programmes mainly consisted of combinations of procedures aiming at the reduction of stress,

the provision of auditory, tactile, visual or vestibular stimuli and passive motor handling proce-

dures. Amongst the programmes which started after discharge from the NICU, Neurodevelop-

mental Treatment (NDT), which consists of a mix of general sensory stimulation and passive and

active motor intervention strategies, was the intervention most frequently used. Other frequently

applied forms of intervention were various forms of developmental programmes, which always in-

cluded general sensory stimulation and general stimulation of motor development, but also could

imply passive handling techniques and the enhancement of parent infant interaction.

Information about the period of application of the intervention was supplied in 30 studies.

Most NICU interventions were applied during variable periods, as most interventions took place

between the age of some postnatal days till discharge. The application period in the post-NICU

studies varied between 2 months and more than 4 years. Also the intensity of intervention showed

considerable heterogeneity. It ranged from once a month to continuous intervention. Programmes

starting after discharge from the NICU were mostly applied within a home based setting. Another

frequently used way to supply intervention was the use of hospital or centre based intervention

combined with home care. In 24 out of the 34 studies parents were incorporated into the interven-

tion. In 19 studies the focus was on enhancing parental skills, in the other five studies the parents

carried out (a part of) the intervention, i.e. the parents functioned as a therapist.

General evaluation of effects (Tables 3.3-3.7)

Most studies evaluated the effect of intervention on motor performance during the intervention,

immediately after the end of intervention and/or some months or 1-2 years after intervention. Var-

ious sorts of outcome measures were used for effect evaluation. We categorized the measures into

46 47

Stud

y N

E

C

Attr

itio

n

(%)

Stud

y gr

oup

Evid

ence

leve

l

Inte

rnal

valid

ity

Exte

rnal

valid

ity

NIC

UA

ls e

t al.

1994

3820

180

HR

I++

++R

esni

ck e

t al.

1987

/198

825

510

711

413

HR

I+

+D

arra

h et

al.

1994

107

5354

51H

RI

++

Ari

agno

et a

l. 19

9735

1414

20H

RI

++

Cha

rpak

et a

l. 20

0174

638

236

416

HR

I+

+N

elso

n et

al.

2001

3721

1630

HR

or

CN

S in

jury

I+

+K

lebe

rg e

t al.

2002

2511

920

HR

I+

+Te

ssie

r et

al.

2003

431

183

153

22H

RI

++

Wes

trup

et a

l. 20

0441

2120

37H

RI

+-

Kor

ner

et a

l. 19

8356

128

64H

RI

--

Feld

man

et a

l. 20

0214

673

739

HR

II-

-H

elde

rs e

t al.

1989

149

6782

34H

RII

--

Als

et a

l. 19

8616

88

0H

RII

I+

-Le

ib e

t al.

1980

2814

140

HR

III

--

Mou

radi

an a

nd A

ls 19

9440

2020

0H

RII

I-

-B

ecke

r et

al.

1999

38?

?61

HR

III

--

Kle

berg

et a

l. 20

0033

1518

?H

RII

I-

-

aft

er N

ICU

– 9

mo

Goo

dman

et a

l. 19

85

8040

4001

HR

I++

++Pi

per

et a

l. 19

8613

466

6814

HR

I++

++Le

kscu

lcha

i and

Col

e 20

0184

4341

14H

RI

+++

Bar

rera

et a

l. 19

8680

3248

26H

RI

+++

Wei

ndlin

g et

al.

1996

105

5154

21H

R o

f CP

I++

+R

othb

erg

et a

l. 19

9149

2821

391

HR

I+

+D

’ Avi

gnon

et a

l. 19

8132

12/1

028

6H

RI

+-

Kan

da e

t al.

2004

105

50

CP

III

--

9 m

o –

18 m

oU

lric

h et

al.

2001

3015

150

DS

I++

++Pa

lmer

et a

l. 19

88/1

990

4825

232

CP

I++

++R

eddi

houg

h et

al.

1998

6632

349

CP

I+

+M

ahon

ey 2

001

5028

220

CP/

DS

II++

+Ei

ckm

ann

et a

l. 20

0315

678

7813

Del

ayed

cog

n/ m

ot d

evII

++

Pipe

r an

d Pl

ess

1980

3721

160

DS

III

+-

aft

er N

ICU

– 1

8 m

oM

ayo

1991

2917

120

Susp

ecte

d C

PI

+++

Sche

rzer

et a

l. 19

7624

148

8C

PI

+++

Har

ris

1981

2010

100

DS

I+

+

tab

le 3

.3. G

roup

s an

d de

sign

.

C =

Con

trol

/con

tras

t gro

up, C

NS

= C

entr

al N

ervo

us S

yste

m, C

P =

cere

bral

pal

sy, D

elay

ed c

ogn

/mot

dev

= D

elay

ed c

ogn

itiv

e/m

otor

dev

elop

men

t,

DS

= D

own

synd

rom

e, E

= E

xper

imen

tal g

roup

, HR

= H

igh

risk

1 In t

he t

ime

betw

een

the

two

stud

ies

31 c

hild

ren

wer

e lo

st to

follo

w-u

p 2 Te

n in

fant

s re

ceiv

ed a

diff

eren

t kin

d of

inte

rven

tion

48 49

neuromotor ones or as tests which provide a more general description of the child’s developmental

level (Table 3.7). The Bayley Scales of Infant Development were most frequently used as outcome

measure for both neuromotor (PDI) and developmental (MDI) outcome. In 26 studies develop-

mental tests were used to evaluate the effect of intervention. In nine of these a beneficial effect of

intervention on the developmental parameters was reported. In 26 studies neuromotor tests were

used. In 13 of these, experimental infants had a better neuromotor outcome than control infants.

Most studies were designed as randomised controlled trials. Twenty-three of the 34 studies

had the highest level of evidence, i.e. level I according to Sackett (see Table 3.1), four studies had a

grade II level of evidence and seven studies were classified as level III. Internal validity was high in

11 studies, fair in 15 and low in eight studies. External validity was in general moderate only: in five

studies generalization was plausible, 17 studies offered some possibilities for generalization and

12 studies had low external validity. Validity of post-NICU studies was usually better than that of

NICU studies.

In the next paragraphs we report the effects of intervention on motor development for the dif-

ferent age periods during which intervention had started, while taking into account the methodo-

logical strength of the studies.

Effect of intervention starting in the NICU

From the 17 NICU studies, eight had a high methodological quality. These studies had an evidence

level I, a fair to high internal validity and they provided at least some possibilities for generaliza-

tion. In two of these eight studies a significantly positive effect of intervention on motor outcome

could be demonstrated. One of the positive studies dealt with the effect of NIDCAP (Als et al

1994). The focus of intervention in this study was stress reduction in combination with general

sensory stimulation. The other intervention aimed at improving the infant’s general developmen-

tal level by means of a developmental programme, including general sensory stimulation, general

stimulation of motor development, passive handling techniques and the enhancement of parent

infant interaction (Resnick et al 1987, 1988). In the other six NICU studies of high methodologi-

cal quality, intervention had no statistically significant effect on motor development (Darrah et

al 1994, Nelson et al 2001, Ariagno et al 1997, Charpak et al 2001, Kleberg et al 2002, Tessier et al

2003). The interventions used in these studies all included procedures to reduce the infant’s level of

stress and multimodal sensory stimulation (either specific or general), which was or was not com-

bined with passive motor intervention techniques or the facilitation of parent infant interaction.

Five of the nine NICU studies with a lower methodological quality pointed to a positive effect of

intervention (Als et al 1986, Mouradian and Als 1994, Feldman et al 2002, Korner et al 1983, Leib et al

1980). Intervention used in these studies consisted of various combinations of procedures to reduce

stress, multimodal sensory stimulation, passive motor intervention strategies or the facilitation of

parent infant interaction. The remaining four studies were unable to demonstrate a beneficial effect

of intervention (Kleberg et al 2000, Westrup et al 2004, Helders et al 1989, Becker et al 1999).

Effect of intervention starting between NICU-discharge and 9 months

Six of the eight studies in which intervention started in the period between discharge from the

NICU and the corrected age of 9 months had a high methodological quality. In four studies,

amongst them the two methodologically strongest studies, the effects of NDT were evaluated

(Goodman et al 1985, Piper et al 1986, Rothberg et al 1991, Weindling et al 1996). None of the

NDT-studies demonstrated a statistically significant effect of intervention on motor develop-

ment. It should be noted however, that in two studies (Goodman et al 1985, Rothberg et al 1991)

outcome was evaluated only by means of global developmental tests. Two other studies (Barrera et

al 1986, Leksculchai and Cole 2001) applied a programme to stimulate infant motor development.

Both programmes consisted of general sensory stimulation and general stimulation of motor

development. In addition, the intervention in the Barrera et al. study included enhancement of

parent infant interaction, that of Leksculchai and Cole passive handling techniques. Both studies

reported a statistically significant positive effect of intervention on motor development. Two stud-

ies addressing the effect of intervention starting in this age period were of limited methodological

quality. One study evaluated in relatively small groups the effect of NDT and that of treatment

according to Vojta (d’Avignon et al 1981). It concluded that outcome in the two treatment groups

did not show a statistically significant difference. The other study reported a dose-response effect

of treatment according to Vojta on motor outcome (Kanda et al 2004).

Effect of intervention starting between 9 and 18 months

Three of the six post-NICU studies, in which intervention started between 9 and 18 months of

age, had a high methodological quality. One study demonstrated a positive effect of specific mo-

tor training on motor development of infants with Down syndrome (Ulrich et al 2001), another

revealed that a general infant stimulation programme facilitated motor development more than

NDT did (Palmer et al 1988, 1990). The third did not find a significant effect of conductive educa-

tion on motor development (Reddihough et al 1998). The other three late post-NICU studies had

a more limited methodological quality. One evaluated the effect of NDT and did not demonstrate

a significant positive effect of intervention (Mahoney 2001). The other two used general pro-

grammes to stimulate motor development. One of them found a beneficial effect of intervention

(Eickmann et al 2003).

Effect of intervention starting between NICU-discharge and 18 months

The three studies in which intervention started between discharge from the NICU and the age

of 18 months were all level I studies with a moderate to high internal validity and a moderate

external validity. Two studies evaluated the effect of NDT. The study which assessed the effect of

a short period of intensive NDT was unable to demonstrate a significant effect of intervention on

motor development (Harris 1981), the other which applied less intensive NDT for half a year did re-

port a positive effect of intervention on motor development (Mayo 1991). The third study evaluated

the effect of a general physical therapy programme and did not find a significant beneficial effect

of the intervention on motor development (Scherzer et al 1976).

50 51

tabl

e 3.

4. T

ype

of in

terv

enti

on.

ATVV = Auditory-Tactile-Visual-Vestibular Stimulation, C = Control group, Ce = Centre, CE = Conduc-tive Education, d = day, Descr. = description, Dev hand = Developmental Handling, Dev Int = Develop-mental Intervention, Dev mile = Developmental milestones, Dev pgm = Developmental programme, Dev S = Developmental Skills, Dev stim = Developmental stimulation, DPI = Developmental Program Intervention, E = Experimental group, h = hour, Hm = home, Hp = Hospital, I = Instruction, IS = Infant

Stud

yM

etho

dD

escr

.Pe

riod

of a

ppli

cati

onIn

ten

sity

Loc

atio

nPa

rent

sO

nse

tE

nd

NIC

U

A

ls e

t al.

1994

NID

CA

P++

3 d

afte

r bi

rth

?C

onti

nuou

sly

Hp

PR

esn

ick

et a

l. 19

87/1

988

Dev

Int

++B

irth

24 m

oH

p: d

aily

H

m: 2

x/m

oH

p/H

mP

Dar

rah

et a

l. 19

94W

b+

2-7

d af

ter

birt

hPl

acem

ent i

n op

en c

ots

Con

tinu

ousl

yH

p?

Ari

agno

et a

l. 19

97N

IDC

AP

+?

?C

onti

nuou

sly

Hp

P

Cha

rpak

et a

l. 20

01K

C+

3-5

d af

ter

birt

hPo

siti

on n

o lo

nger

acc

epte

dC

onti

nuou

sly

Hp

P

Nel

son

et a

l. 20

01AT

VV

++33

wk

PMA

2 m

oH

p: 1

5 m

in, 2

x/d

5x/w

k ,

Hm

: 2x/

d H

p/H

mT

Kle

berg

et a

l. 20

02N

IDC

AP

+1 d

aft

er b

irth

? C

onti

nuou

sly

Hp

PTe

ssie

r et

al.

2003

KC

++co

ndit

ion

stab

le

Dis

char

geC

onti

nuou

sly

Hp

PW

estr

up e

t al.

2004

NID

CA

P+

1 d a

fter

bir

th36

wk

PMA

Con

tinu

ousl

yH

pP

Kor

ner

et a

l. 19

83W

b+

< 4

d af

ter

birt

hT

ill e

valu

atio

nC

onti

nuou

sly

Hp

?

Feld

man

et a

l. 20

02K

C++

31-3

4 w

k PM

AD

isch

arge

, du

rati

on ≥

14d

> 1 h

/dH

pP

Hel

ders

et a

l. 19

89T

sti

m/ R

F-

Bir

thD

isch

arge

?H

p?

Als

et a

l. 19

86N

IDC

AP

++9

d af

ter

birt

hD

isch

arge

Con

tinu

ousl

yH

pP

Lei

b et

al.

1980

Sen

s En

r+

Bir

thD

isch

arge

?H

p?

Mou

radi

an a

nd A

ls

1994

NID

CA

P+

?<

40 w

PM

AC

onti

nuou

sly

Hp

P

Bec

ker

et a

l. 19

99D

ev h

and

+B

irth

36 w

k PM

A?

Hp

?K

lebe

rg e

t al.

2000

NID

CA

P+

3 d

afte

r bi

rth

36 w

k PM

A?

Hp

P

aft

er N

ICU

– 9

mo

Goo

dman

et a

l. 19

85N

DT

-3

mo

12 m

oI:

1x/

mo

> 45

min

P:

dai

ly (h

ome

prog

ram

me)

Hp/

Hm

P

Pipe

r et

al.

1986

ND

T+

Term

age

12 m

oI:

0-3

mo:

1x/

wk

3-

12 m

o: 1

x/2w

kP:

dai

lyH

mP

Lek

scul

chai

and

Col

e 20

01D

ev p

gm++

Term

age

4 m

oI :

1x/

mo

P: d

aily

Hm

T

Bar

rera

et a

l. 19

86D

PI /

PIT

+4

mo

16 m

oI:

4-7

mo:

1-2

h/w

k

7-13

mo:

1x/

2wk

13

-16

mo:

1x/

mo

Hm

P

Wei

ndli

ng e

t al.

1996

ND

T-

Term

age

12 m

o (C

P

con

tinu

e)I:

0-6

mo:

1x/

wk

6-

9 m

o: 1

x/2w

k

9-

12 m

o: 1

x/m

o

Hm

?

Rot

hber

g et

al.

1991

ND

T+

3 m

o12

mo

I: 1

x/m

o >

45 m

in

P: d

aily

(h

ome

prog

ram

me)

Hp/

Hm

P

D’ A

vign

on e

t al.

1981

Vojt

a/N

DT

-4-

7 m

oB

: > 3

mo

V: >

6 m

o ?

??

Kan

da e

t al.

2004

Vojt

a-

1 mo

E: M

ean

52 m

oC

: Mea

n 25

mo

E: ≥

30

min

, 3-

4x/d

ayH

mT

9 m

o –

18 m

oU

lric

h et

al.

2001

TT

+9-

12 m

o In

depe

nden

t w

alki

ng8

min

/d, 5

d/w

k H

mT

Palm

er e

t al.

1988

/199

0N

DT/

IS++

12-1

9 m

o D

urat

ion

12 m

oI:

1 h

1x/2

wk

P: d

aily

(h

ome

prog

ram

me)

Ce/

Hm

P

Red

diho

ugh

et a

l. 19

98C

E++

12-3

6 m

oD

urat

ion

6 m

oE:

2.8

h/w

k C

: 2.9

h/w

kC

e?

Mah

oney

200

1N

DT/

Dev

S+

Mea

n ag

e 14

mo

Dur

atio

n 12

mo

3x/m

o, 4

5 m

inC

e/H

m?

Eick

man

n et

al.

2003

Dev

sti

m+

13 m

o18

mo

I: 1

1 hm

vis

its

30-4

5 m

in,

3 w

orks

hops

Hm

P

Pipe

r an

d Pl

ess

1980

Dev

mile

+M

ean

age

9 m

oD

urat

ion

6 m

oI:

1 h

1x/2

wk

P: h

ome

prog

ram

me

Ce/

Hm

P

aft

er N

ICU

– 18

mo

May

o 19

91N

DT

+4-

18 m

oD

urat

ion

6 m

oE:

1 h/

wk

C: 1

h/m

o H

p/H

mP

Sche

rzer

et a

l 197

6Ph

ys T

+5-

17 m

o24

mo

1h 1

x/2w

kH

mT

Har

ris

1981

ND

T++

2-21

mo

Dur

atio

n 9

wk

E: 3

x/w

k >4

0 m

inH

m?

Stimulation, KC = Kangaroo Care, min = minutes, mo = months (corrected age), NDT = Neurodevelopmental Treatment, NIDCAP = Newborn Individualized Developmental Care and Assessment Program, P = parents, P= enhance parental skills, PIT = Parent-Infant treatment, PMA = post menstrual age, Phys T = Physical Therapy, Sens. Enr. = Sensory Enrichment, T = parents are ‘therapist’, T stim/RF = Tactile Stimulation/ Range Finding, TT = Treadmill Training, Wb = Waterbed, wk = weeks, ? = no information available

52 53

tab

le 3

.5. E

valu

atio

n of

eff

ect o

f int

erve

ntio

n.

age

s of

eva

luat

ion

Out

com

e m

easu

res1

res

ults

2

<ter

mte

rm1-

9 m

o9-

18 m

o>1

8 m

o N

euro

mot

orD

evel

opm

enta

l

NIC

UA

ls e

t al.

1994

--

19

-A

PIB

, Bay

ley

PDI

Bay

ley

MD

IE

> C

Res

nic

k et

al.

1987

/198

8-

--

1224

Bay

ley

PDI

Bay

ley

MD

IE

> C

Dar

rah

et a

l. 19

941x

+4,

812

18M

AI

Peab

ody

E =

CA

riag

no e

t al.

1997

1x-

1,4

1224

Bay

ley

PDI,

NA

PI, A

PIB

Bay

ley

MD

IE

= C

Cha

rpak

et a

l. 20

01-

+3,

69,

12-

-G

riffi

thE

= C

Nel

son

et a

l. 20

01-

-2,

412

-B

ayle

y PD

IB

ayle

y M

DI

E =

CK

lebe

rg e

t al.

2002

--

-12

-B

ayle

y PD

IB

ayle

y M

DI

E =

CTe

ssie

r et

al.

2003

--

-12

--

Gri

ffith

E =

CW

estr

up e

t al.

2004

--

--

66M

ovem

ent A

BC

-E

= C

Kor

ner

et a

l. 19

831x

--

--

LA

PPI

-E

> C

Feld

man

et a

l. 20

021x

-3,

6-

-B

ayle

y PD

IB

ayle

y M

DI

E >

CH

elde

rs e

t al.

1989

pre,

aft

er 2

wk,

wee

kly

till

disc

harg

e-

-PM

Dev

E =

CA

ls e

t al.

1986

4x+

1,3,

69

-A

PIB

, Bay

ley

PDI

Bay

ley

MD

IE

> C

Lei

b et

al.

1980

2x-

6-

-B

ayle

y PD

I, N

BA

SB

ayle

y M

DI

E >

CM

oura

dian

and

Als

1994

--

1-

-A

PIB

-E

> C

Bec

ker

et a

l. 19

993x

--

--

VA-

E =

CK

lebe

rg e

t al.

2000

--

--

36-

Gri

ffith

E =

C

aft

er N

ICU

– 9

mo

Goo

dman

et a

l. 19

85

--

69,

12-

-G

riffi

thE

= C

Pipe

r et

al.

1986

--

612

-W

olan

ski

Mil

ani,

Gri

ffith

E =

C

Leks

culc

hai a

nd C

ole

2001

-+

1,2,

3,4

--

TIM

P-

E >

CB

arre

ra e

t al.

1986

--

416

-B

ayle

y PD

IB

ayle

y M

DI

E >

CW

eind

ling

et a

l. 19

96-

--

1230

MA

IG

riffi

thE

= C

Rot

hber

g et

al.

1991

--

--

72-

Gri

ffith

-II

E =

CD

’Avi

gnon

et a

l. 19

81-

--

-36

-72

CP

clas

s-

E =

CK

anda

et a

l. 20

04-

-1,

2,3,

5ev

ery

3 m

o 59

Neu

rEx

-E

> C

9 m

o –

18 m

oU

lric

h et

al.

2001

--

ever

y 2

wk

Bay

ley

PDI

Bay

ley

MD

IE

> C

Palm

er e

t al.

1988

/199

0-

--

-18

,24

Bay

ley

PDI

Bay

ley

MD

IE

< C

Red

diho

ugh

et a

l. 19

98-

-pr

e-po

stG

MFM

-E

= C

Mah

oney

200

1-

--

-14

,26

-B

ayle

y M

DI,

Peab

ody

E =

C

Eick

man

n et

al.

2003

--

-12

18B

ayle

y PD

IB

ayle

y M

DI

E >

CPi

per

and

Ples

s 19

80-

--

pre-

post

-G

riffi

thE

= C

aft

er N

ICU

– 18

mo

May

o 19

91-

-pr

epo

stW

olan

ski

Bay

ley

MD

I,Ges

ell

E >

C

Sche

rzer

et a

l 197

6-

-pr

e24

MD

CG

esel

lE

= C

Har

ris

1981

--

pre

post

Bay

ley

PDI

Bay

ley

MD

I, Pe

abod

yE

= C

C =

Con

trol

gro

up, E

= E

xper

imen

tal g

roup

, mo

= m

onth

s, M

P =

mot

or p

erfo

rman

ce, p

re =

bef

ore

inte

rven

tion

, pos

t = a

fter

inte

rven

tion

, wk

= w

eeks

1 For

expl

anat

ion

of a

bbre

viat

ion

s se

e T

able

3.7

2 St

atis

tica

lly s

ign

ifica

nt d

iffer

ence

s fo

und

in m

otor

out

com

e at

old

est a

ge o

f eva

luat

ion:

E >

C:

Exp

erim

enta

l gro

up s

ign

ifica

ntly

bet

ter

outc

ome

than

C

ontr

ol G

roup

, E =

C: n

o di

ffer

ence

bet

wee

n gr

oups

, E <

C:

Con

trol

Gro

up b

ette

r ou

tcom

e th

an E

xper

imen

tal G

roup

.

54 55

Intervention Sensory Stimulation

Motor Intervention

Enhancing PII

S. r. S. u. S. m. G. m. P. a. s. a. g.Waterbed + - + - + - - -ATVV intervention + - + - - - - -NIDCAP + - - + + - - +KC + - - + + - - +Developmental handling + - - + + - - ?Treadmill training - + - - - + - -Tactile stimulation/Range finding - + - - + - - -Vojta - + - - + - - -Sensory enrichment - - + - - - - -Developmental intervention - - + - + - + +NDT - - - + + - + -Developmental program - - - + + - + -Developmental milestones - - - + - - + +Developmental Stimulation - - - + - - + +Developmental Parent intervention - - - + - - + +Infant Stimulation - - - + - - + +Conductive Education - - - + - - + -Developmental skills - - - + - - + ?Parent Infant treatment - - - - - - - +

+ = procedure used, - = procedure not used,? = insufficient information available,

PII = Parent Infant Interaction; for other abbreviations see legends Table 3.4.

S. R. = Stress Reduction, S. u. = Specific –unimodal, S. m. = Specific – multimodal, G. m. = General – multimo-

dal, P. = Passive, A. s. = Active – specific, A. g. = Active – general

Stress reduction: decreasing stressful events to the body by restricting input from the environment until the

infant is capable of maintaining an adequate organization of its behavioural state; placing the infant in a way as

to provide a sense of containment similar to the intrauterine environment.

Sensory stimulation:

a) specific-unimodal: procedures during which a single sensory modality is stimulated (e.g., specific tactile

stimulation)

b) specific-multimodal: procedures during which multiple specific sensory modalities are stimulated (e.g.,

ATVV which consists of the application of auditory, tactile, visual and vestibular stimuli)

c) general-multimodal: procedures during which multiple forms of not explicitly described sensory stimuli are

applied (e.g., verbal and tactile encouragement as part of general developmental programmes)

table 3.6. Composition of programmes used in EI. Motor intervention strategies:

a) passive procedures: the therapist or the parent carries out specific techniques, which do not require active

motor behaviour of the child, i.e., the child has a passive role (e.g. handling, positioning, and facilitation

procedures)

b) active-specific: the child is encouraged to actively train a specific motor ability (e.g., walking by means of

treadmill training)

c) active-general: the child is encouraged to train a variety of motor abilities; stimulation of activities occurs

in general by means of structured activities which are designed to meet the child’s developmental level.

Practice and play are important elements in the latter type of intervention.

Enhancing parent-infant interaction: parents receive information on infant behaviour. The increased knowl-

edge on infantile behaviour facilitates parent’s sensitivity to the child’s needs and promotes developmentally

supportive behaviour.

Discussion

We are not the first to write a review on the effects of early intervention for children at high risk

for developmental disabilities, others preceded us: Simeonsson et al 1982, Parette and Hourcade

1984, Casto and Mastropieri 1986, Shonkoff and Hauser-Cram 1987, Parry 1992, Turnbull 1993,

Guralnick 1997, Majnemer 1998. The major conclusion from these reviews is that the evidence

favouring EI is inconclusive. The results from this present review indicate that we have moved a

bit forward. In the following paragraphs we will point out the direction of progress. But before we

address the issues of ‘which programme at what age’, we first discuss some methodological issues.

Methodological considerations

The studies included in this review were very heterogenic in nature. Not only a large variation

existed in the number of subjects included in the studies, but also in the intervention methods

which started prior to 18 months. The interventions themselves, the outcome measures used

to evaluate the intervention methods and the ages at which outcome was determined were too

heterogeneous to allow for a formal meta-analysis. The methods of intervention in the NICU

period varied from NIDCAP and Kangaroo Care to different kinds of stimulation programmes.

In post NICU studies NDT was the leading method of intervention, but there were also studies

which used interventions like infant stimulation, conductive education and other developmental

programmes.

It was encouraging to notice that 20 out of 34 studies had a high methodological quality, i.e.,

they had an evidence level I and a fair to high internal and external validity. This reflects that

during recent years the requirement of good quality studies on the effect of early intervention has

been increasingly materialized (Siebes et al 2002). Previous studies indicated that with an increase

in the rigorousness of the studies the support for effectiveness of EI decreases (Parette and Hour-

cade 1984, Siebes et al 2002). For the studies included in the present review this also holds true.

Of the 20 studies with a high methodological quality only six (30%) were able to demonstrate a sig-

56 57

Neu

rom

otor

test

Dev

elop

men

tal t

est

Bay

ley

Scal

es o

f In

fant

Dev

elop

men

t -

Psyc

hom

otor

Dev

elop

men

t Ind

ex

Bay

ley-

PDI

1969

19

93G

riffi

ths

Dev

elop

men

tal S

cale

s G

riffi

th19

5419

70

Wol

ansk

i Gro

ss M

otor

Eva

luat

ion

Wol

ansk

i19

73M

ilan

i-C

ompa

rett

i Mot

or

Dev

elop

men

t Scr

een

ing

test

M

ilan

i19

67

Neo

nat

al B

ehav

iora

l Ass

essm

ent

Scor

e N

BA

S19

73

1984

Bay

ley

Scal

es o

f In

fant

Dev

elop

men

t -

Men

tal D

evel

opm

ent I

ndex

B

ayle

y-M

DI

1969

19

93M

otor

Dev

elop

men

t Che

ckli

st

MD

C

1976

Ges

ell D

evel

opm

enta

l Sch

edul

es

Ges

ell

1974

Mov

emen

t Ass

essm

ent o

f In

fant

s M

AI

1980

Peab

ody

Dev

elop

men

tal M

otor

Sca

les

Peab

ody

1974

Ass

essm

ent o

f Pre

term

Infa

nts’

Beh

avio

r A

PIB

1982

Mue

nche

ner

Fun

ktio

nelle

En

twic

klun

gs D

iagn

osti

kM

FED

1978

Lon

gitu

din

al n

euro

beha

vior

al a

sses

smen

t

proc

edur

e fo

r pr

eter

m in

fant

sL

APP

I19

83Ps

ycho

mot

or D

evel

opm

ent p

rofi

le

(bas

ed o

n G

esel

l)PM

Dev

1981

Supp

lem

enta

l Mot

or T

est f

or P

ostu

ral C

ontr

ol

PoC

o19

87N

euro

beha

vior

al A

sses

smen

t of t

he P

rete

rmIn

fant

NA

PI19

90

Mov

emen

t Ass

essm

ent B

atte

ry fo

r C

hild

ren

Mov

emen

t A

BC

1992

Gro

ss M

otor

Fun

ctio

n M

easu

re

GM

FM19

93Te

st o

f In

fant

Mot

or P

erfo

rman

ce

TIM

P19

95

Vid

eo-a

nal

ysis

(am

ount

and

var

iati

on in

mov

e-m

ents

)VA

1999

Cli

nic

al N

euro

logi

cal e

xam

inat

ion

Cli

n N

eur

Ex

n.f.

s.*

CP

clas

sific

atio

nC

P cl

ass

1981

*not

furt

her

spec

ified

tabl

e 3.

7. C

lass

ifica

tion

of o

utco

me

mea

sure

s in

to n

euro

mot

or te

sts

and

deve

lopm

enta

l tes

ts.

nificant beneficial effect of intervention on motor development. From the 14 studies with a limited

methodological quality, seven (50%) reported a positive effect of intervention.

Studies on the effect of intervention in children with or at risk for developmental disorders -

like the ones included in the present review - are often hampered by specific problems (Majnemer

1998, Siebes et al 2002). First, many studies include small study groups with a large heterogeneity

of degree and type of problems thereby diminishing generalizability and statistical power. Second,

assigning participants to a control group which does not receive treatment usually is considered

unethical. Therefore, the results of most intervention studies represent only the additional value

of the intervention under study. Another significant problem concerns the existing standard-

ized outcome measures. In general they are characterized by a lack of sensitivity to detect small

changes in motor development although these small changes may have an important influence

on the functional abilities of the child. The studies included in this review not only used a large

variation in outcome variables (see Table 3.7), but the measures were also mainly discriminative.

The use of discriminative measures, which focus on the comparison of a child’s score with an

age-equivalent score, may be one of the reasons that so little effect of EI is found. Another problem

associated with the used outcome measures is that they mainly measure quantitative changes in

motor development instead of qualitative ones and measures which focus on changes in func-

tional abilities (Ketelaar et al 1998).

Few studies addressed the effect of intervention on outcome beyond preschool age. This

means that we lack information on the effect of EI on the child’s activities of daily life and the

child’s socialization brought about by the potentially beneficial effect of EI on motor development.

Future studies should address the effect of EI on these outcome parameters, as they have a major

impact on the child’s participation in society.

The results of our review are discussed while taking age at the onset of intervention as a pri-

mary focus. The number of studies with a high methodological quality in the various age periods

after NICU discharge was so low that it precluded conclusions on the effect of age at onset of

intervention after term age. We therefore decided to discuss the results of our review in two sec-

tions, one for NICU studies and one for post-NICU studies.

NICU studies

Eight of the 17 NICU studies had a high methodological quality. Three of those evaluated the

effects of NIDCAP intervention. One demonstrated a significant positive effect on motor develop-

ment as measured by the psychomotor index of the Bayley Scales of Infant Development (Bayley –

PDI; Als et al 1994), but the other two were unable to reveal such an effect on Bayley – PDI (Kleberg

et al 2002, Ariagno et al 1997). The difference in outcome between the three studies might be

attributed to the age at which outcome was assessed.

Outcome in the positive effect study of Als et al (1994) was evaluated at 9 months corrected

age, in the two ‘no effect’ studies at 12 and 24 months. Thus, it could be that NIDCAP has a tem-

porary beneficial effect on motor development of infants at high risk for developmental disorder.

58 59

This notion is in line with results of two recent meta-analyses which concluded that NIDCAP

has a temporary beneficial effect on cognitive and motor development (Jacobs et al 2002, Sym-

ington and Pinelli 2003). But it should be kept in mind that only the study of Westrup et al (2004)

assessed the effect of NIDCAP beyond the age of two years. The latter small study was unable to

demonstrate a significant positive effect of NIDCAP on developmental outcome at the age of 5½

years. Nevertheless, considering reports that NIDCAP intervention in low risk preterm infants

has a significant positive effect on electrophysiological and MRI correlates of brain development

at 42 weeks postmenstrual age (Buehler et al 1995, Als et al 2004), it is conceivable that NIDCAP

might affect complex motor behaviour and cognitive abilities at school age. It might be that this

putatively positive effect will be found in particular in low risk preterm infants and not in high

risk preterms.

Two other high quality studies used Kangaroo Care to improve motor outcome (Charpak et

al 2001, Tessier et al 2003). The application of Kangaroo Care had no effect on developmental

outcome as measured by the Griffiths Developmental Scales at 6 and 12 months corrected age. Two

explanations for this result can be offered. First, it could be that Kangaroo Care does not affect

motor development. It is likely that the effect of the relatively simple Kangaroo Care is weaker

than that of the rather complex NIDCAP programme. Second, it is possible that the effects are too

subtle to be caught by the Griffiths scales.

Two high quality studies applied intervention strategies consisting of procedures to reduce

stress in combination with specific multimodal sensory stimulation with or without passive mo-

tor intervention procedures (Darrah et al 1994, Nelson et al 2001). Both studies were unable to find

a positive effect of intervention on motor development at 12 and 18 months corrected age.

The last high quality NICU study showed that developmental intervention had a significant

positive effect on motor development (Resnick et al 1987, 1988). In this programme intervention

started in the hospital with vestibular and visual stimulation to promote development. After dis-

charge, intervention was continued for two years by means of a developmental programme in the

home situation. The parents played an important role in carrying out the intervention programme

which consisted of 400 different motor, social and cognitive activities complemented by several

parenting activities. The positive outcome of the intervention probably can be attributed more to

the duration of the programme and the continuous involvement of the parents in the development

of their children than by the NICU part of the intervention.

In conclusion, the current review provides little evidence that intervention during the NICU

period in infants at high risk for developmental disorders has a beneficial effect on motor develop-

ment. Yet, a potential advantageous effect of NIDCAP on motor development cannot be excluded.

We recommend that further studies address the effect of NIDCAP on developmental outcome at

school age in low risk and high risk preterm infants.

Post NICU studies

From the 17 studies which started after the NICU period, 12 had a high methodological qual-

ity. Only four of those were able to show a beneficial effect of intervention on motor develop-

ment. Eight of the 12 studies evaluated the effects of NDT or physiotherapy mainly based on the

principles of NDT. It is striking that only one of these studies reported a better motor outcome in

the experimental group than in the control group (Mayo 1991). The positive effect study of Mayo

differed from the other studies, by being the only study which compared intensive NDT treatment

(once a week) to less intensive NDT (once a month). The other eight studies compared NDT to in-

fant stimulation (Palmer et al 1988, 1990) or to a not further defined form of standard care. In six

of the seven studies motor outcome in the NDT group was similar to that of the contrast group.

In the seventh study motor development was worse in children treated according to the principles

of NDT than in children who received an infant stimulation programme (Palmer et al 1988, 1990).

The above studies indicate that NDT during the first years of life does not have a measurable

positive effect on motor development. This is in line with the conclusion of a recent review on the

effects of NDT for people with cerebral palsy, aged 5 months to 22 years, that NDT did not have a

clear beneficial effect on developmental outcome (Butler and Darrah 2001).

The other four high quality studies evaluated the effects of a developmental programme, treadmill

training or conductive education. The two developmental programme studies (Barrera et al 1986,

Leksculchai and Cole 2001) and the treadmill training study (Ulrich et al 2001) reported a positive

effect of intervention on motor development. The fourth study compared the effect of conductive

education with that of traditional neurodevelopmental programmes. Both types of intervention

were associated with similar degrees of developmental progress (Reddihough et al 1998).

Treatment according to Vojta was only evaluated in two studies. D’Avignon and colleagues

(1981) compared in a small randomised trial with a limited methodological quality the effect of

treatment according to Vojta with NDT. They reported that the groups did not differ significantly

in developmental outcome. Kanda et al (2004), who studied the effect of the amount of Vojta

treatment on developmental outcome, reported a better outcome for the group which received suf-

ficient Vojta training compared to the group which had had insufficient Vojta therapy. However, a

major drawback of the study is that the design suffered from self-selection of the groups.

In conclusion, the current review indicates that intervention programmes in the first post-

natal years according to the principles of NDT or Vojta do not have a beneficial effect on motor

development in children at high risk for developmental disorders or children with CP or Down

syndrome. However, substantial evidence has been provided that specific developmental training

and general developmental programmes in which parents learn how to promote infant develop-

ment can exert a positive effect on motor development.

60 61

Concluding remarks

The present review indicates that intervention in children at risk of developmental disabilities

should be adapted to the infant’s age, i.e. the type of intervention which might be beneficial for

infants at preterm age differs from the type which is effective in infants who have reached at least

term age.

At preterm age infants seem to benefit most from intervention which aims at mimicking the

intrauterine environment, such as the NIDCAP intervention. Some evidence has been provided

that NIDCAP might have a temporary beneficial effect on infant motor (and cognitive) devel-

opment. Future studies should address the question whether NIDCAP affects developmental

outcome at school age.

The studies conducted after term indicated that intervention programmes according to the

principles of NDT or Vojta, i.e., programmes in which passive handling techniques play a promi-

nent role, do not have a clear beneficial effect on motor development. But intervention by means of

specific motor training programmes, such as training of locomotor movements on a treadmill and

general developmental programmes, where intervention aims at stimulation of the child’s explora-

tion of active motor behaviour, can exert a positive effect on motor development. Too few of these

training and developmental programme studies were available to answer the question whether

the age at which intervention starts matters, leaving this question for future research.

acknowledgements

We gratefully acknowledge the critical comments of Prof. Dr. B.F. van der Meulen, Drs. V.B.

de Graaf-Peters, Ms. T. Dirks on a previous version of the manuscript. CHB-H was financially

supported by the Johanna KinderFonds, Stichting Fonds de Gavere and the Graduate School for

Behavioural and Cognitive Neurosciences (BCN).

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65

C H a P t E r 4

d oe s e A r ly

I n t e rV e n t Ion I n

I n fA n t s At h Ig h r I s k

f or A de V e l oPm e n tA l

mo tor dI s or de r

I m P ro V e mo tor

A n d c o g n I t I V e

de V e l oPm e n t ? ---------------------------------------------------------------------------------------------------

CH Blauw-Hospers, VB de Graaf-Peters, T Dirks, AF Bos, M Hadders-Algra

Neuroscience and Biobehavioral Reviews, 2007, 31(8); 1201-1212

66 67

abstract

Infants at high risk for developmental motor disorders are in general referred to early interven-

tion (EI) services. It is a matter of debate to which extent EI may facilitate outcome in various

developmental domains. We reviewed the effects of EI programmes aiming at promoting motor

and cognitive development. With respect to motor development the data indicated that EI prior to

term age probably is most effective when it aims at mimicking the intrauterine environment; after

term age general developmental programmes probably are most effective. Some evidence was pro-

vided that EI prior to term age has a beneficial effect on cognitive development regardless the type

of intervention which is applied. After term age only general developmental programmes seemed

to have an effect on cognitive development. The review concludes with preliminary data on the

effect a new intervention programme, COPCA, applied between 3 and 6 months corrected age on

developmental outcome till 18 months. The results indicated that COPCA was more beneficial for

the development of sitting behaviour and cognition than traditional paediatric physiotherapy.

Keywords

Early intervention, motor development, cognitive development, developmental disorder, infant,

COPCA

Introduction

Infants who are born with a low birth weight or preterm are at biological risk for developmental

disorders (Aylward et al 1989, Bhutta et al 2002). Therefore these infants often are called high

risk (HR) infants. In general HR infants have poorer cognitive, motor and behavioural outcomes

than infants born at term. The less advantageous outcome is not only present at school-age, but

persists into adolescence and adulthood (Bhutta et al 2002, Marlow 2004, Johnson et al 2005). The

developmental outcome of infants born prematurely is heterogeneous and can be partly associ-

ated with biological risk factors, such as gestational age, birth weight, brain damage acquired in

the pre- or perinatal period or subsequent illnesses. However, these factors account only for a

portion of the variance associated with long term outcomes. In part the outcome is also mediated

by environmental experience (Shonkoff and Meisels 2000, Vohr et al 2000, Aylward 2005, Johnson

et al 2005, Spittle et al 2005). The complex character of influences on development makes it almost

impossible to determine the relation between biological risk, environmental risk and neurodevel-

opmental outcome. Another factor which influences developmental outcome is the so-called plas-

ticity of the brain. Depending on the timing of the insult to the brain and the size and site of the

lesion functional recovery may occur. The brain is plastic in particular during the phase occurring

after the completion of neuronal migration, during which the processes of dendritic outgrowth

and synapse formation are highly active (Kolb et al 2001). The latter means that considerable

plasticity can be expected between 2-3 months before until about 12-15 months after term age (de

Graaf-Peters and Hadders-Algra 2006).

While major disorders such as mental retardation, epilepsy or cerebral palsy – which occur

in 6-25% of HR infants (Aylward 2005) – are often identified during infancy, the effects of minor

neurological dysfunction (MND) become more obvious with increasing age and are for instance

expressed in learning disorders, attention deficit hyperactivity disorder (ADHD), clumsiness or

other behavioural problems. MND occurs in as many as 50-70% of HR infants (Aylward 2005). Two

basic forms of MND have been distinguished, i.e. simple and complex MND (Hadders-Algra 2002).

Simple MND occurs relatively frequently and it is most probably caused by genetic predisposition

and stress during pre- or perinatal life. Simple MND might be considered to represent the less

optimal but still normal brain function. Complex MND is strongly related to perinatal adversities.

It might be considered as a borderline form of CP and shows a clear association with motor, cogni-

tive and behavioural problems (Hadders-Algra 2002, 2003).

The variety in developmental problems that may occur as a result of pre- and perinatal adver-

sities makes it difficult to recognize infants at high risk for both major and minor neurological

dysfunction at early age. The development of imaging techniques has improved the possibilities to

identify these infants at early age (Aylward 2005). However, these techniques are not always able

to visualize abnormalities in the brain (Hadders-Algra 2000b). Another instrument which might

assist the early identification of HR infants is the assessment of the quality of General Movements

(GMs) (Prechtl 2001, Hadders-Algra 2004). GMs are complex movement patterns involving head,

trunk, arms and legs. They are present from early foetal life and disappear around 3-4 months

68 69

post term when goal-directed motor behaviour emerges. The quality of GMs, which is primarily

determined by movement complexity and variation, reflects the integrity of the nervous system.

Various studies indicate that the presence of definitely abnormal GMs around 3 months post-term,

i.e. in the last phase of GM-development, is associated with a high risk for developmental motor

disorders, such as CP (Prechtl et al 1997, Prechtl 2001, Hadders-Algra 2004, Groen et al 2005).

The early detection of infants at high risk for developmental disorders offers the opportunity

for intervention at young age, i.e. during a phase in which the central nervous system is character-

ized by considerable plasticity. It is however still a matter of debate to what extent early interven-

tion (EI) may facilitate outcome in various developmental domains. Therefore the first aim of the

present paper is to review the effects of EI programmes which aim primarily at the improvement

of motor development on a) motor and b) cognitive outcome. The analyses were restricted to in-

terventions starting prior to 18 months corrected age. The third part of the paper summarizes the

first results of an ongoing EI-study in which the effect of a recently developed physiotherapeutic

intervention programme for infants with high risk for developmental disorders is compared to the

effect of EI based on the principles of NeuroDevelopmental Treatment (NDT).

Effects of EI on motor development of Hr infants

Recently we reviewed the literature on the effects of EI on motor development of HR infants

(Blauw-Hospers and Hadders-Algra 2005). We first will summarize the findings of this review.

This will be followed by a discussion and integration of additional studies which have subsequent-

ly been published. We started with a literature search which covered various databases, including

Medline, CINAHL, PsycINFO and PEDro, in the period 1966 – June 2004. Thirty four studies

were identified which fulfilled the following four criteria: a) subjects of study: infants with high

biological risk for or with developmental disabilities, b) aim of the intervention (mostly amongst

others): to improve motor development, c) onset of intervention in at least 50% of the participating

children: between birth and the (corrected) age of 18 months, d) journal in which the study was

published had an impact factor of > 0.3. Excluded were studies restricted to medical and orthopae-

dic interventions and studies in populations of healthy low-risk preterm infants or in populations

of socially disadvantaged children without specific biological risk for developmental disorders.

The data revealed that a large diversity of EI programmes is applied. Before term age New-

born Individualized Developmental Care and Assessment Program (NIDCAP; Als et al 1994) is

mostly used, after term age a whole range of specific and general developmental programmes are

applied, together with more traditional physiotherapeutic interventions such as NeuroDevelop-

mental Treatment (NDT; Bobath 1980, Howle 2002) and treatment according to Vojta (Vojta 1976).

Specific developmental programmes aim at the improvement of one aspect of motor development

for example walking skills, while general developmental programmes aim at improvement in all

developmental domains. Parents usually play an important role in the implementation of the

specific and general developmental programmes.

The methodological quality (MQ) of each study was analysed systematically by taking into ac-

count 1) the level of evidence (Sackett 1989), 2) internal and 3) external validity. It was encouraging

to note that 20 out of 34 studies had a high MQ, i.e. they had an evidence level I and a fair to high

internal and external validity. This indicates a considerable improvement in the quality of studies

evaluating the effect of early intervention (Siebes et al 2002). Previous studies indicated that with

an increase in the rigorousness of the studies the evidence for the effectiveness of EI decreases

(Parette and Hourcade 1984, Siebes et al 2002). For the studies included in the present review this

also held true. Of the 20 studies with a high methodological quality only six (30%) showed a sig-

nificant beneficial effect of intervention on motor development. From the 14 studies with a limited

methodological quality, seven (50%) reported a positive effect of intervention.

The review indicated that intervention which might be beneficial for infants at preterm age is

different from the type which is effective in infants who have reached at least term age. At preterm

age infants seem to benefit most from intervention which aims at mimicking the intrauterine

environment, such as NIDCAP intervention. After term age, intervention by means of specific or

general developmental programmes has a positive effect on motor development. No evidence was

found for a beneficial effect of traditional forms of paediatric physiotherapy like NDT and Vojta.

An additional literature search was performed to cover the period July 2004 – September

2006. The results of this search are shown in Table 4.1. Another five studies were identified which

fulfilled the four aforementioned selection criteria. In two studies the onset of intervention was

during the period of stay at the Neonatal Intensive Care Unit (NICU; Oghi et al 2004, Cameron

et al 2005). Both studies had a fair methodological quality (MQ), consisting of a combination of

level I evidence (Sackett 1989), a fair internal validity, and a limited external validity. The stud-

ies applied different intervention techniques in different groups of children and were unable to

demonstrate a beneficial effect of intervention on motor development. The other three studies

started between discharge from the NICU and the age of 9 months (Koldewijn et al 2005, Badr et

al 2006, Gianni et al 2006). Gianni et al (2006) evaluated the effects of a mother-child intervention.

The study had a fair methodological quality and was unable to demonstrate a beneficial effect of

intervention on motor development. The other studies had a lower MQ. Badr et al (2006) studied

the effects of a cognitive/sensorimotor stimulation programme on motor development, but was not

able to demonstrate an effect of the intervention. The study of Koldewijn et al (2005) reported the

results of a pilot study on the Infant Behavioral Assessment and Intervention Program (IBAIP). They

demonstrated that at both 3 and 6 months motor development improved significantly compared to

the control group. But due to the fact that they had used a historical control group the methodologi-

cal quality of this study was limited (evidence level III, fair internal but low external validity).

We conclude that the five recent studies on the effect of early intervention in infants at high

risk for developmental motor disorders do not alter the conclusions of the original review (Blauw-

Hospers and Hadders-Algra 2005): intervention prior to term age probably is most effective when

it aims at mimicking the intrauterine environment; intervention after term age probably is most

effective when it consists of stimulation of motor development by means of the promotion of self-

produced motor behaviour.

70 71

tabl

e 4.

1. C

hara

cter

isti

cs o

f add

itio

nal

stu

dies

(Jul

y 20

04-S

epte

mbe

r 20

06) o

n th

e eff

ect o

f EI o

n m

otor

dev

elop

men

t.

Stud

y ch

arac

teri

stic

s

Stud

y N

EC

Attr

itio

n(%

)St

udy

grou

p Ev

iden

cele

vel

Inte

rnal

valid

ity

Exte

rnal

valid

ity

NIC

UO

hgi e

t al.

2004

2412

12

4H

R w

ith

CN

S in

jury

I+

-C

amer

on e

t al.

2005

7234

3817

HR

I++

+

aft

er N

ICU

– 9

mo

Kol

dew

ijn e

t al.

2005

4424

2011

HR

III

(his

tori

cal

cont

rol)

+-

Gia

nn

i et a

l. 20

0636

1818

0H

RI

+++

Bad

r et

al.

2006

6232

3031

HR

wit

h C

NS

inju

ryI

+-

Pro

gram

me

char

acte

rist

ics

Stud

yM

etho

dD

escr

.Ap

plic

atio

n pe

riod

Inte

nsit

yLo

cati

onPa

rent

s

NIC

UO

hgi e

t al.

2004

EIP

++pr

ior

to d

isch

arge

-6

mo

prio

r to

dis

char

ge:

3-4x

30

min

.A

fter

: 1x

wk/

2w

k 40

-60

min

Hos

pita

l/C

ente

rP

Cam

eron

et a

l. 20

05N

eon

atal

Dev

pg

m, l

arge

ly

base

d on

ND

T

++bi

rth

- 4 m

o - h

ospi

tal d

aily

- dep

endi

ng o

n ri

sk m

ax

1x 2

wk

Hos

pita

l /

Hom

eP

aft

er N

ICU

– 9

mo

Kol

dew

ijn e

t al.

2005

IBA

IP++

1 wk

afte

r di

scha

rge

- 6 m

o3x

eva

luat

ion,

6-8

hom

e in

terv

enti

ons

Hom

eP

Gia

nn

i et a

l. 20

06M

othe

r-ch

ild

inte

rven

tion

+3

mo

- 12

mo

2x p

er m

onth

, 1.5

hC

ente

rP

Bad

r et

al.

2006

Cog

nit

ive-

sen

sori

mot

or

stim

ulat

ion

prog

ram

+di

scha

rge

- 12

mo

1 mo:

2x

wk,

til

l 4 m

o:

1x w

k,th

erea

fter

1x

2wk

Hom

eP

C =

Con

trol

gro

up, C

NS

= C

entr

al N

ervo

us S

yste

m, E

= E

xper

imen

tal g

roup

, EIP

= E

arly

Inte

rven

tion

Pro

gram

, h =

hou

r, H

R =

Hig

h ri

sk,

IBA

IP =

Infa

nt B

ehav

iora

l Ass

essm

ent a

nd In

terv

enti

on P

rogr

am, m

in =

min

utes

, mo

= m

onth

s (c

orre

cted

age

), n

= nu

mbe

r, N

DT

= N

euro

de-

velo

pmen

tal T

reat

men

t, N

eon

atal

Dev

pgm

= N

eon

atal

Dev

elop

men

tal p

rogr

am, P

= p

aren

ts, P

= en

hanc

e pa

rent

al s

kills

, T =

par

ents

are

‘th

erap

ist’,

wk

= w

eeks

, ? =

no

info

rmat

ion

avai

labl

eO

utco

me

mea

sure

s: A

IMS

= A

lber

ta In

fant

Mot

or S

cale

, Gri

ffith

= G

riffi

ths

Dev

elop

men

tal S

cale

s, IB

A =

,MD

I = B

ayle

y Sc

ales

of I

nfa

nt D

e-ve

lopm

ent –

Men

tal D

evel

opm

ent I

ndex

, NB

AS

= N

eon

atal

Beh

avio

ral A

sses

smen

t Sco

re B

ayle

y, B

ayle

y PD

I = B

ayle

y Sc

ales

of I

nfa

nt D

evel

op-

men

t – P

sych

omot

or D

evel

opm

ent I

ndex

, 1 St

atis

tica

lly s

ign

ifica

nt d

iffer

ence

s fo

und

in m

otor

out

com

e at

old

est a

ge o

f eva

luat

ion:

E >

C:

Exp

erim

enta

l gro

up s

ign

ifica

ntly

bet

ter

out-

com

e th

an C

ontr

ol G

roup

, E =

C: n

o di

ffer

ence

bet

wee

n gr

oups

, E <

C:

Con

trol

Gro

up b

ette

r ou

tcom

e th

an E

xper

imen

tal G

roup

.

The

tab

le s

how

s th

e ch

arac

teri

stic

s of

stu

dies

on

earl

y in

terv

enti

on fo

cusi

ng o

n m

otor

dev

elop

men

t pub

lishe

d be

twee

n Ju

ly

2004

and

Oct

ober

200

6. In

the

tab

le ‘s

tudy

cha

ract

eris

tics

’ the

sam

ple

size

, stu

dy g

roup

and

cri

teri

a fo

r th

e m

etho

dolo

gica

l qu

alit

y ar

e pr

esen

ted.

The

sec

ond

part

‘pro

gram

me

char

acte

rist

ics’

desc

ribe

s th

e m

etho

ds o

f int

erve

ntio

n to

geth

er w

ith

the

dura

tion

and

inte

nsi

ty o

f tre

atm

ent,

the

loca

tion

whe

re t

he t

reat

men

t was

app

lied

and

the

role

of t

he p

aren

ts in

the

inte

rven

-ti

on p

rogr

amm

e. In

the

thi

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72 73

Do EI programmes which focus on motor development affect cognitive outcome?

It is generally acknowledged that HR infants, as a group, have lower cognitive scores than typically

developing infants and that their cognitive abilities may deteriorate over time (Vohr and Garcia

Coll 1985, Aylward 1989, Bhutta et al 2002, Ment et al 2003). Therefore early intervention pro-

grammes often do not only aim at the improvement of motor outcome but also at the facilitation

of cognitive development.

Over the years the effects of various general developmental intervention programmes aiming

at the improvement of cognitive development have been studied extensively for both environmen-

tally and biologically disadvantaged infants. Studies on early intervention by means of general

developmental programmes in children who are environmentally disadvantaged indicate both a

short and long term beneficial effect on cognitive development (Ramey and Campbell 1984, Wasik

et al 1990, Campbell and Ramey 1994). The programmes designed for infants at biological risk

for developmental disorders show less consistent effects of EI by means of general developmental

programmes on cognitive development. For instance, the Vermont Intervention Program found

both short and long term effects on cognitive function (Rauh et al 1988, Achenbach et al 1990,

Achenbach et al 1993). Studies on the Infant Health and Development Program (IHDP) however

indicated that immediately after the intervention at the age of three years a positive effect on

cognitive development was present (Infant Health and Development Program 1990), but that

the effect disappeared in the follow up at 5 and 18 years for the group with the higher biological

risk status, i.e. the infants with a gestational age at birth of less than 37 weeks and a birth weight

of less than 2000 grams (Brooks-Gunn et al 1994, McCormick et al 2006). In the children with

the lower biological risk, i.e. who had a birth weight between 2001 and 2499 grams, the effect

of intervention was still visible at the age of 18 years. The findings of the IHDP programme are

consistent with the results of a more recent study on a general developmental programme, the

Avon Premature Infant Project, which showed that the beneficial effect of the intervention which

was observed at two years (Avon Premature Infant Project 1998) had disappeared when the study

group was re-examined at five years of age (Johnson et al 2005). Thus general developmental pro-

grammes aiming at the improvement of cognitive development are indeed successful.

We wondered whether EI programmes focussing primarily on motor development have a

similar effect on cognitive outcome. The hypothesis underlying this assumption is that improve-

ment of early motor development allows the infant more opportunities to interact with the

environment, which in turn may facilitate cognitive development (Becker 1999). To this end we re-

examined whether the EI programmes which primarily aimed to improve motor development and

were included in the review described in the previous section also evaluated cognitive outcome.

To ensure the completeness of our study we carried out an additional literature search covering

different databases including MEDLINE (1966 to November 2006), CINAHL (1982 to November

2006), AMED (1985 to November 2006), PsycINFO (1967 to November 2006) and PEDro. The

keyword ‘early intervention’ was searched in combination with one of the following keywords:

‘infant’, ‘motor development’, ‘cognitive development’, ‘low birth weight’, ‘preterm’, ‘high-risk’,

‘developmental disabilities’ and ‘cerebral palsy’. We used the same inclusion and exclusion criteria

as in the review on motor development, be it that in the current review both motor and cognitive

outcome measures had to be included. The search did not yield other studies than those already

identified for the previous review. Twenty six studies of the previous 34 studies included in the

review also had a cognitive outcome measure. The results of the effect of EI on cognitive develop-

ment are summarized in Table 4.2.

In 13 of the 26 identified studies intervention had started in the NICU period, in eight studies

intervention was initiated in the period between discharge from the NICU and the corrected age

of 9 months and in three studies intervention started between 9 and 18 months. In two other

studies the preset age criteria were not fulfilled i.e. they started at some age between discharge

from the NICU and 18 months. All studies were reviewed on methodological quality (MQ). A fair

MQ was present in sixteen studies. They all evaluated cognitive outcome with either the men-

tal scale of the Bayley Scales of Infant Development (BSID) or with the Griffiths Developmental

Scales.

Seven studies with a fair MQ evaluated outcome of intervention which had started during the

NICU period. Five out of these seven studies reported a beneficial effect of intervention on cogni-

tive development (Resnick et al 1987, 1988, Als et al 1994, Nelson et al 2001, Kleberg et al 2002,

Tessier et al 2003). Interestingly the five studies evaluated four different types of early interven-

tion programmes: Developmental Program including stimulation, NIDCAP, Auditory-Tactile-

Visual-Vestibular Stimulation, and Kangaroo Care. The other six studies had a lower MQ. Three

suggested a positive effect on cognitive development (Leib et al 1980, Als et al 1986, Feldman et al

2002). Six of the eight studies that started after discharge from the NICU but before the corrected

age of nine months had a fair MQ. Two demonstrated that EI had a beneficial effect on cognitive

outcome (Barrera et al 1986, Gianni et al 2006). Both studies evaluated the effects of a general

developmental programme. The other four studies with fair MQ which did not find a positive

effect of EI on cognitive development all addressed the effects of NeuroDevelopmental Treatment

(Goodman et al 1985, Piper et al 1986, Rothberg et al 1991, Weindling et al 1996).

74 75

StudyEvidence Level1

Internal validity2

External validity3

Results

motor4

Results cognitive4

NICUAls et al. 1994 I ++ ++ E > C E > CResnick et al. 1987/1988 I + + E > C E > CAriagno et al. 1997 I + + E = C E = CCharpak et al. 2001 I + + E = C E = CNelson et al. 2001 I + + E = C E > CKleberg et al. 2002 I + + E = C E > CTessier et al. 2003 I + + E = C E > CWestrup et al. 2004 I + - E = C E = COhgi et al 2004 I + - E = C E = CFeldman et al. 2002 II - - E > C E > CAls et al. 1986 III + - E > C E > CLeib et al. 1980 III - - E > C E > CKleberg et al. 2000 III - - E = C E = C

after NICU – 9 moGoodman et al. 1985 I ++ ++ E = C E = CPiper et al. 1986 I ++ ++ E = C E = CBarrera et al. 1986 I ++ + E > C E > CWeindling et al. 1996 I ++ + E = C E = CRothberg et al. 1991 I + + E = C E = CGianni et al. 2006 I ++ + E = C E > CBadr et al. 2006 I + - E = C E = CKoldewijn et al. 2005 III + - E > C E > C

9 mo – 18 moPalmer et al. 1988/1990 I ++ ++ E < C E = CEickmann et al. 2003 II + + E > C E > CPiper and Pless 1980 III + - E = C E = C

after NICU – 18 moMayo 1991 I ++ + E > C E > CHarris 1981 I + + E = C E = C

table 4.2. Effect of early intervention on cognitive development.

Only one of the three studies in which intervention started between 9 and 18 months had a fair

MQ (Palmer et al 1988, 1990). This study demonstrated a beneficial effect of an infant stimulation

programme on motor development, but the programme did not affect cognitive outcome. Both

studies in which intervention started between discharge from the NICU and 18 months had a fair

MQ. The study of Mayo (1991) on the effect of NDT demonstrated a significant improvement in

cognitive function for the group which received the intensive treatment approach. The study of

Harris (1981) addressed the effect of NDT in children with Down syndrome; it failed to demon-

strate a positive effect on cognitive development.

In conclusion, the review provides some evidence that intervention during the NICU period

in infants at high risk for developmental motor disorders has a beneficial effect on cognitive devel-

opment. Interestingly, the effect seems to be independent of the type of intervention. The studies

in which intervention had started after term age indicated that intervention by means of a general

developmental programme might be beneficial for cognitive development. This finding is consist-

ent with the results of studies on the effect of EI in infants with high biological risk for develop-

mental disorders which primarily aim at improving cognitive outcome. Our review indicated that

NDT applied during infancy does not produce benefits in the cognitive domain – a finding in line

with an earlier review of Butler and Darrah (2001).

the early intervention project in Groningen (VIP project)

The review above indicates that convincing evidence for a beneficial effect of physiotherapy

programmes most frequently used in early intervention (NDT and treatment according to Vojta)

is lacking. This inspired two members of the Groningen team (TD and MHA) to develop a new

physiotherapeutic EI programme: COPCA (Coping with and Caring for infants with neurological

dysfunction – a family centred programme). COPCA is based on the principles of the Neuronal

Group Selection Theory (NGST; Edelman 1993) and on new insights in the field of education and

family care (Dale 1996, Rosenbaum et al 1998). According to NGST typical development is charac-

terized by two phases of variability, i.e. primary and secondary variability. During primary vari-

Table lay-out is based on Blauw-Hospers and Hadders-Algra 2005.1 Evidence level according to Sackett’s method for grading research (Sackett 1989):

I = Randomised controlled trials; II = Non-randomised controlled trials / Prospective cohort studies with concurrent control group; III = Cohort studies with historical control group / Case studies with control participants

2 Internal validity is determined by various study variations such as subject assignment, study design, attri-tion, confounders, validity of outcome measures, blind assessment

3 External validity is determined by possibilities for generalization (group size and internal validity)4 Statistically significant differences found in motor outcome at oldest age of evaluation:

E > C: Experimental group significantly better outcome than control group E = C: No difference between groups E < C: Control group better outcome than experimental group

76 77

ability the nervous system explores all motor possibilities available. In typically developing infants

this phase is characterized by abundant variation. At function specific ages the infant reaches the

phase of secondary variability. The child gradually learns to select the most efficient solution for a

given task out of his motor repertoire. This selection is based on trial and error. This means that

during the phase of secondary variability the child learns to adapt his/her motor behaviour to

specific situations (Hadders-Algra 2000a). Infants with a brain lesion resulting in a developmental

motor disorder have a reduced repertoire of motor strategies available for exploration. In addition,

these infants have problems with the selection of the most appropriate solution for a certain task

out of the repertoire (Hadders-Algra 2000a). Therefore COPCA aims to promote variation in mo-

tor behaviour and trial-and-error experiences, with the ultimate goal that the child will be able to

find an appropriate solution for any motor task.

An equally important base of COPCA are new insights in the field of education and family

care (Dale 1996, Law et al 1998, Rosenbaum et al 1998). Family centred care implies a partnership

between the family and the professional, in which the family defines the priorities for interven-

tion, while the therapist assists the family throughout the intervention period (Law et al 1998). In

COPCA the physiotherapist (PT) acts as a coach who respects the autonomy of the family and in-

tends to make him- or herself superfluous. Caregivers are key-persons in the COPCA programme.

They determine the goals to be achieved. The PT provides information on infant development, the

specific problems of infants with neurological dysfunction, basic principles on infant education,

and specific ways in which development may be promoted and problems may be tackled. By means

of a continuous dialogue with the PT, caregivers thus develop their own ways to cope with their

infant.

In the so-called VIP project (VIP is the abbreviation of the Dutch term for Early Intervention

Project, namely Vroegtijdige Interventie Project) the effect of COPCA on developmental outcome

till the age of 18 months is currently studied. Here we will present preliminary data on the first

20 infants included in the project who have already reached the age of 18 months. The aim of the

study is to assess whether COPCA is more beneficial for developmental outcome at 18 months cor-

rected age than traditional paediatric physiotherapy (TPP), which in the Netherlands in general

consists of the implementation of the ‘living concept’ of NDT. The latter implies that TPP is

practised with significant heterogeneity.

The study groups of the VIP project consisted of infants who had been admitted to the NICU

of the University Medical Center Groningen (UMCG) between March 2003 and May 2005 and

showed at ten weeks corrected age definitely abnormal GMs. As indicated in the introduction the

presence of definitely abnormal GMs indicates a high risk for developmental motor disabilities,

such as cerebral palsy (CP) (Prechtl 2001, Hadders-Algra 2004). Infants with severe congenital

anomalies and infants whose parents had an inappropriate understanding of the Dutch language

were excluded from the study. All parents of the infants gave informed consent and the project

was approved by the Ethics Committee of the University Medical Center Groningen.

The infants were randomly assigned to the COPCA or the control group. The randomised

intervention period lasted from 3 to 6 months corrected age1. Thereafter the intervention followed

the suggestions of the paediatrician in charge of the care of the infant. COPCA intervention was

provided twice a week for one hour in the home environment by one of four specially trained

paediatric physiotherapists. The control group received TPP with a frequency varying from 2 to 20

times (median value: 7) during the intervention period. The duration of the TPP sessions varied

from 15 to 36 minutes (median value: 30); in general they were also applied in the home environ-

ment. During the study the assessors were blind regarding group allocation.

Of the first twenty infants enrolled in the VIP project nine had been assigned to COPCA in-

tervention and 11 to the control group. The latter got physiotherapeutic guidance indicated by the

paediatrician. It turned out that nine infants in the control group had received TPP and two got

no specific guidance. As the results of the two infants who did not receive TPP did not differ from

those who did, we decided to include these two infants without TPP in the control group. The

COPCA and control group were similar in terms of gender distribution, gestational age at birth

and birth weight (Table 4.3). All but two infants had been born preterm.

table 4.3. Clinical characteristics of the two study groups.

COPCa (n = 9) tPP (n = 11)

Gender 4 male

5 female

5 male

6 female

Preterm (GA<37 wk) n = 8 n = 10

Term n = 1 n = 1

Gestational age (wk) 27-37

(median 29)

27-39

(median 31)

Birth weight (g) 585-3640

(median 1160)

825-3460

(median 1340)

1 Throughout the rest of the document ages in months are ages corrected for preterm birth.

78 79

The infants’ developmental status was assessed with a battery of tests at 3 months (baseline), 4, 5,

6 months and 18 months. At each age the assessment included a standardized neurological exami-

nation according to Prechtl (1977) with age-specific adaptations of the norms according to Touwen

(1976) or at 18 months according to Hempel (1993) and the Alberta Infant Motor Scale (AIMS; Piper

and Darrah 1994). The neurological findings were summarized as normal, minor neurological

dysfunction (MND) or abnormal. During early infancy abnormal indicated the presence of a full-

blown neurological syndrome, such as a clear hypo- or hypertonia, a hemisyndrome, or a hyperex-

citability syndrome; at 18 months it denoted the presence of CP.

MND indicated the presence of signs of neurological dysfunction not meeting the criteria

for clear neurological pathology. The signs of MND were grouped into the following functional

clusters: mild abnormalities in gross motor function, mild abnormalities in fine motor function,

mild abnormalities in muscle tone regulation, mildly abnormal reflexes, and mild cranial nerve

dysfunction (Hadders-Algra 2003). Age specific criteria for deviancy in a specific cluster were

determined. On the basis of the number of clusters meeting the criteria for deviancy two forms of

MND could be distinguished: simple MND, which denoted the presence of one cluster of dysfunc-

tion and complex MND, when more than one cluster fulfilled the criteria for deviancy (Hadders-

Algra 2002, 2003). At the age of 18 months we also applied the optimality concept to summarize

neurological condition (Huisman et al 1995). For 57 items, which represent the neurological

examination, criteria for optimality were defined. It is important to realize that the definition of

optimal is narrower than that of normal or typical. The Neurological Optimality Score (NOS) of

an infant is the sum of items meeting the criteria for optimality, which can count up to a maxi-

mum of 57 points.

The AIMS (Piper and Darrah 1994) was used to evaluate gross motor development in various

positions (supine, prone, sitting and standing) over time. The 58 items each describe three aspects

of motor performance – weight-bearing, posture and antigravity movements. The assessment

results in a total score and subscores for the various positions. At 6 and 18 months also the Mental

Scale of Bayley Scales of Infant Development (BSID-II; Bayley 1993, Van der Meulen et al 2002) was

administered. The mental scale consists of items concerning discrimination, classification, language,

social skills, memory and problem solving. The raw scores of the mental scale of the Bayley were con-

verted into age-equivalent scores, as derived from the Dutch norms (Van der Meulen et al 2002).

The results showed that both groups did not differ in neurological condition. This was true for

baseline, for short term outcome at 6 months and for long term outcome at 18 months. At 18

months, two children in each group were diagnosed with CP. All other infants showed the com-

plex form of MND (Figure 4.1). As an important part of the neurological examination consists of

the assessment of muscle tone, reflexes and cranial nerve function, it could perhaps be that both

types of intervention have a similar effect on these neural functions. Therefore, we also analyzed

neurological condition at 18 months by means of the neurological optimality score. The neurolog-

ical optimality score focuses on the quality of motor behaviour in terms of variability and fluency.

The analysis of the optimality scores was applied only in the children without CP. The results sugges-

ted a small, but statistically not significant advantage for the infants of the COPCA group (Figure 4.2).

Figure 4.1. Neurological condition of infants of the COPCA and TPP group at 3, 6 and 18 months. The numbers refer to the actual number of infants.

2

4

2

2

2

2

1

7

3 mo

COPCa

tPP

3 mo

6 mo

6 mo

18 mo

18 mo7

6

9

7

9

= Simple MND = Complex MND = Abnormal / CP

Neu

rolo

gica

l Opt

imal

ity

scor

e

COPCa(N = 7)

COPCatPP

tPP(N = 9)

p = 0.3140

35

30

25

20

Figure 4.2. Effect of type of intervention on the neurological optimality score at 18 months in children without cerebral palsy. Vertical bars, range; horizontal lines, median value; boxes: interquartile range. Mann-Whitney U test: p=0.31.

80 81

tota

l aIM

S sc

ore

COPCatPP

21

18

15

12

6

61

91

9

3 4 6

Figure 4.3. Effect of type of intervention on the total AIMS score at 3, 4 and 6 months. Vertical bars, range; horizontal lines, median value; boxes: interquartile range.

The effect of intervention on the development of gross motor skills was analyzed with the AIMS.

The data revealed that at the age of 18 months the AIMS suffers from a ceiling effect (Darrah et al

1998, Liao and Campbell 2004). We therefore restricted our analyses to the data of 3 to 6 months.

The analyses were carried out twice, once for all infants and once without the children who devel-

oped CP. The rationale behind the second series of analyses was that it is conceivable that children

with mild to moderate dysfunction profit more from intervention than children with severe dys-

function. The analyses showed no differences in total AIMS scores between the COPCA and the

control group at baseline, during and immediately after the intervention period (Figure 4.3). Nei-

ther did the groups differ in scores on the subscales which assess motor behaviour in supine, prone

and standing position. On the sitting subscale however a significant positive effect of the COPCA

intervention was observed whether or not the infants who developed CP were included into the

analysis (p=0.03 with CP included and p=0.02 with CP excluded; Figure 4.4a, 4.4b). Furthermore,

the effect of COPCA intervention tended to increase with increasing age when children with CP

were left out of the analysis (p=0.08) (Figure 4.4b). The results suggest that the infants who re-

ceived COPCA intervention have a better postural control at 6 months than the infants of the TPP

group. These findings match with the results of de Graaf-Peters et al (2006), who demonstrated by

means of EMG recordings that the infants who received COPCA intervention showed at 6 months

a more typical pattern of postural control during sitting than the infants of the TPP group.

Figure 4.4 Effect of type of intervention on the sitting subscale of the AIMS at 3, 4, and 6 mo: a) with infants with CP included, b) with infants with CP excluded. Vertical bars, range; horizontal lines, median value; boxes: interquartile range. MANOVA: main effect of intervention * p=0.03, ** p=0.02. Intervention and age tended to interact (p=0.08).

Finally, the results showed that COPCA intervention might also have an effect on cognitive

development. At 6 months the MDI of both groups was identical. In the TPP group we found a

significant decrease in the median MDI score over time from 109 at 6 months to 99 at 18 months

(p=0.03). In general it is known that cognitive abilities of preterm infants deteriorate with increas-

ing age (Vohr and Garcia Coll 1985, Aylward 1989, Bhutta et al 2002, Ment et al 2003). In contrast

to the TPP group, the MDI score of the infants who had received COPCA intervention remained

stable over time (median values at 6 months 110, at 18 months 111; Figure 4.5). This indicates that

COPCA intervention might be able to counteract a cognitive deterioration.

Sitt

ing

subs

cale

Sitt

ing

subs

cale

COPCattP

a. B.

1

3

55 21

17

21

98 98

49 49

34 46 6

1

2 2

3 3

4 4

5 5

82 83

Concluding remarks

Prior to term age intervention which aims at mimicking the intrauterine environment is probably

most effect for motor development; for cognitive development any type of intervention seems to

be beneficial. After term age EI probably is most effective for motor and cognitive development

when a general developmental programme is applied. No evidence is available supporting the

notion that intervention according to the principles of NDT has a positive effect on motor or

cognitive development. The limited amount of evidence favouring EI encouraged two members

of the Groningen team (TD and MHA) to develop a new physiotherapeutic intervention, COPCA.

Preliminary results indicate that COPCA has a significant effect on the development of sitting

abilities and that COPCA might be able to counteract a cognitive deterioration. However, further

research is needed to explore clinical effectiveness of the COPCA programme.

acknowledgements

We kindly acknowledge the contribution of our collaborators Kune Blaauw PT, Lily van Doormaal

MD, Ellen Janssen PT, Peter Logtenberg PT and Wilma Renkema PT MSc. CHB-H was financially

supported by the Johanna KinderFonds, Stichting Fonds de Gavere, the Cornelia Stichting and the

Graduate School for Behavioural and Cognitive Neurosciences (BCN).

Figure 4.5 Bayley Scales of Infant Development: MDI scores at 6 and 18 months in COPCA and TPP group. Vertical bars, range; horizontal lines, median value; boxes: interquartile range. Wilcoxon: * p=0.03.

BSI

D-I

I MD

I

COPCatPP

60

80

100

120

140

6 mo 6 mo18 mo 18 mo

*

COPCa tPP

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C H a P t E r 5

de V e l oPm e n t of A

q uA n t I tAt I V e to ol

to A s s e s s t h e c on t e n t

of P h ys Ic A l t h e r A P y

f or I n fA n t s

------------------------------------------------------------------------------------------------------------

CH Blauw-Hospers, T Dirks, LJ Hulshof, M Hadders-Algra

Pediatric Physical Therapy, 2010; 22: 189-198

88 89

abstract

Purpose. The study aim was to describe and quantify physical therapy interventions for infants at

high risk for developmental disorders.

Methods. An observation protocol was developed based on knowledge about infant physical thera-

py and analysis of directly observable physiotherapeutic (PT) actions. The protocol’s psychometric

quality was assessed. Videos of 42 infant physical therapy sessions at 4 or 6 months corrected age

were analyzed.

results. The observation protocol classified PT actions into 8 mutually exclusive categories.

Virtually all PT actions during treatment could be classified. Interrater and intrarater agreement

were satisfactory (intraclass correlations, 0.68-1.00). Approximately 40% of treatment time was

spent on challenging the infants to produce motor behavior by themselves whereas approximately

30% of time facilitation techniques were applied. Tradition-based sessions could be differentiated

from function-oriented ones.

Conclusions. It is possible to document PT actions during physical therapy treatment of infants at

high risk for cerebral palsy in a systematic, standardized and reliable way.

Key Words

Physical Therapy, Infants, Cerebral Palsy, Program evaluation, Video observation, Quantitative

tool, Reliability

Introduction and purpose

In pediatric physical therapy, multiple intervention programs are used for the treatment of infants

with or at risk for developmental disorders, such as cerebral palsy (CP). Programs frequently used

during a stay in the neonatal intensive care unit are the Newborn Individualized Developmental

Care and Assessment Program, Kangaroo Care and sensory stimulation programs. Programs

used after term age are quite different in approach and vary from treatment according to Vojta, to

NeuroDevelopmental Treatment (NDT), to multiple forms of specific and general developmental

programs (Blauw-Hospers and Hadders-Algra 2005). Four recent reviews (Butler and Darrah 2001,

Blauw-Hospers and Hadders-Algra 2005, Blauw-Hospers et al 2007, Spittle et al 2007) did not find

conclusive evidence that early physical therapy intervention improves motor outcome in children

with or at risk for CP. Literature also does not provide evidence that early physical therapy pro-

duces other benefits, such as the enhancement of social-emotional or cognitive function (Butler

and Darrah 2001, Blauw-Hospers et al 2007).

A “theory driven evaluation” (Chen 1990) might help to understand why evidence for the ef-

fectiveness of early intervention (EI) programs is lacking. Theory driven evaluations pay attention

to the mechanisms underlying outcome. This means that they do not only evaluate the effect of

intervention on outcome (eg, “does application of EI result in a higher score on the Bayley Scales of

Infant Development?”), but also the pathway along which the outcome is achieved (eg, “is applica-

tion of a specific physiotherapeutic (PT) action related to outcome?”).

Theory driven evaluation offers a framework to understand the relation between program

inputs, the contents of the program - the ‘black box’ -, and the outcomes (Brazil et al 2005). Know-

ledge about the effect of specific elements of an intervention is crucial for improvement of physi-

cal therapeutic guidance. Guidance should focus on elements which have a beneficial effect on

outcome and elements which do not affect outcome or have an adverse effect should be discarded.

Uncovering the actual contents of practiced physical therapy will shed light on potentially effec-

tive and ineffective elements of the intervention.

The aim of the present study is to develop an instrument to objectify what physical therapists

actually do during treatment, that is, to open the black box of physical therapy sessions in daily

practice, applied to infants at high risk for a developmental disorder such as CP. It should be noted

that CP develops in only a portion of infants at high risk for developmental disorders. Yet, motor

problems and difficulties in learning develop in a substantial proportion of these infants at school

age (Aylward 2005).

To this end, we developed an observation protocol based on the theoretical constructs of the

most commonly used physical therapy approaches for high risk infants in the Netherlands, that

is, NDT and functional therapy (Ketelaar et al 2001, Ekström et al 2005). The term functional in

the latter approach may give the impression that NDT does not aim at improving the infant’s

function. This is a false impression; all approaches in infant physical therapy aim at improving

the child’s function. Nevertheless, some differences exist between NDT and functional therapy.

For instance, NDT is characterized by a broad repertoire of therapist-infant contact strategies,

90 91

whereas the functional approach relies entirely on infant self-produced motor behavior. Primary

questions addressed in this study deal with the psychometric properties of the protocol. 1) Is the

observation protocol complete, that is, is it possible to classify the majority (> 90%) of the contents

of physical therapy sessions? 2) Are the observation items mutually exclusive? 3) Can the items

be assessed reliably in terms of inter- and intraassessor agreement? Secondary questions address

the actual content of the physical therapy sessions during early infancy: 4) Which observable

therapeutic strategies can be distinguished? 5) What proportion of time is spent on the different

strategies?

Methods

Development of the observation protocol

Protocol development started with a systematic analysis of the literature on the application of

physical therapy in infants with or at risk for developmental motor disorders such as CP. This

analysis gave us clues to the most important strategies that are used by pediatric physical thera-

pists. These strategies are based on the theoretical constructs of the most commonly used physical

therapy programs for high-risk infants, that is, NDT and functional therapy. The NDT approach

was developed in the 1940s by Karl and Berta Bobath. Inhibition of spasticity and facilitation of

normal posture and motor behavior were the main aims of treatment, which the Bobaths tried to

accomplish by tone-influencing patterns with the use of various therapeutic handling techniques

(Bobath and Bobath 1984, Mayston 2001). Over the years, NDT gradually evolved based on new

theoretical constructs and practical knowledge. Although the basic philosophy and the approach

to treatment remained the same, goals, treatment techniques and treatment application obtained

a more functional orientation (Mayston 2001, Howle 2002). Currently the main goals of NDT are

to increase or improve the skill repertoire, to maintain the functional level of performance, and

the general management and minimization of contractures and deformities (Mayston 2001).

The way in which NDT is applied depends on the child’s age and the severity of the disability.

Already in the 1960s Elizabeth Köng and Mary Quinton started to adapt and develop techniques

for early treatment of babies based on the Bobath concept (Bly 1999, Quinton and Nelson 2002).

The principles of NDT baby treatment (Bly 1999) aim at helping the infant’s motor development

by giving the infant sensorimotor experiences of typical motor patterns. This is achieved by using

several forms of facilitation techniques, such as handling or pressure techniques, by using sup-

port devices or by giving sensory experience to the infant. Therapy should not be restricted to the

therapy sessions; the therapist aims by means of training the caregivers to achieve a carry-over

from treatment into activities of daily life (Bobath and Bobath 1984, Finnie 1996, Mayston 2001).

The focus of functional therapy lies on improvement of functional tasks (Ketelaar et al 2001,

Ekström et al 2005). Application of the concept of functional therapy to young infants implies that

the cornerstone of guidance consists of encouragement of self produced motor behavior during

functional tasks, such as feeding, dressing, bathing and playing (Resnick et al 1987, Palmer et al

1988, Ketelaar et al 2001, Ekström et al 2005). From the literature, it is known that infants at high

risk for a developmental motor disorder have a reduced repertoire of motor strategies available for

exploration. This is already expressed during the first postnatal months in a limited repertoire of

general movements (GMs) and continues when goal-directed motility emerges. The infants also

have difficulties in selecting the most appropriate solution for a certain task out of their motor

repertoire (Hadders-Algra 2000b). Functional therapy therefore aims through play to promote

variation in motor behavior and opportunities to explore the motor possibilities to find appropri-

ate solutions for motor tasks (Hadders-Algra 2000b, Ketelaar et al 2001, Damiano 2006). Part and

parcel of the more functional approach is the coaching role of the therapist, that is, the caregivers

determine how developmental strategies highlighted by the therapist may be integrated into the

child and family’s daily life.

In the next stage of protocol development we analyzed 20 pilot video recordings of infant

treatment sessions in terms of directly observable PT actions. All actions that physical therapists

performed were documented. These PT actions were classified and defined into categories. The

categories varied from classic NDT actions such as handling techniques to categories describing

functional activities (self produced motor behavior) and family involvement and education. The

observation protocol is the final result of a cyclical process that incorporated knowledge about

physical therapy for infants and the observation of overt and directly observable therapeutic

actions during treatment from video recordings. During the development of the observation pro-

tocol, we paid attention to required psychometric requirements: completeness, mutual exclusive-

ness, reliability, and construct validity (Reynders 1992).

Subjects

The contents of physical therapy sessions were analyzed for a study group of 22 infants, who had

been admitted to the neonatal intensive care unit of the University Medical Center Groningen. All

infants had an indication for early physiotherapeutic intervention on the basis of the presence of

definitely abnormal GMs at the age of ten weeks corrected age. The GM method is a standardized

technique to assess neurological integrity on the basis of the quality of spontaneous motor behav-

ior (Einspieler et al 2004, Hadders-Algra 2004). GMs are spontaneously generated complex move-

ments involving the head, trunk, arms, and legs. Four classes of GM quality can be distinguished:

normal-optimal, normal-suboptimal, mildly abnormal and definitely abnormal. Various studies

showed that the presence of definitely abnormal GMs at 2 to 4 months post-term indicates a high

risk for developmental disabilities, such as CP (Prechtl et al 1997, Prechtl 2001, Hadders-Algra

2004). The GM assessment has a good predictive validity for both major and minor developmental

disorders. Also construct and concurrent validity and reliability of the GM method are satisfac-

tory (Heineman and Hadders-Algra 2008). Infants with severe congenital anomalies and infants

whose caregivers had an inappropriate understanding of the Dutch language were excluded from

the study. All caregivers of the infants signed an informed consent and the research project was

approved by the Ethics Committee of the University Medical Center Groningen.

Twenty of the infants were born preterm with a gestational age at birth that varied from

92 93

28 to 32 weeks (median, 31) and a birth weight between 630 and 2090 g (median, 1205 g). Two

infants were born at term with a median birth weight of 3560 g. Neurological condition was as-

sessed at 6 months corrected age (CA) with the Touwen Infant Neurological Examination (TINE)

(Hadders-Algra et al 2009). Three infants showed clear neurological dysfunction. Fourteen

infants showed a high number of signs of minor neurological dysfunction (MND). The remaining

five infants had a normal-suboptimal neurological condition at 6 months (Table 5.1).

table 5.1. Birth characteristics and neurological outcome (n=22).

total (n = 22)

Gender 10 boys

12 girls

Gestational age (wk)

Median

Range

Preterm (n=20)

31

28-32

Term (n = 2)

38

37-39

Birth weight (g)

Median

Range

Preterm (n=20)

1205

630-2090 g

Term (n =2)

3560

3460-3660 g

Neurological examination at 6 mo* 4 Normal-suboptimal

15 MND

3 Abnormal

* corrected for prematurity

g = gram, MND = Minor Neurological Dysfunction, mo = months, wk = weeks

Recording of intervention sessions

All 22 infants received physical therapy between the ages of 3 to 6 months corrected age. Therapy

was delivered by 17 physical therapists who were registered in the Netherlands as pediatric physi-

cal therapists. Fifteen therapists treated the infants in their home environment and 2 applied the

intervention in a clinical setting. Treatment duration varied from 12 to 50 minutes per session

(mean, 30 minutes per session; SD 9 minutes per session).

At 4 and 6 months corrected age, a video recording of a treatment session of each infant was

made. To minimize intrusion, the camera was positioned as far away from the physical therapist

and infant as possible. In addition, therapist and caregiver were asked to ignore the person ma-

king the video so that the recording would resemble a natural situation. Research has shown that

video recording hardly affects the behavior of people that are being filmed because they tend to

forget the presence of the camera after awhile (Albrecht et al 2005). Care was taken that physical

therapist, caregiver, and infant were continuously in view of the camera.

The video recordings were analyzed with the standardized observation protocol with help of

the Noldus software program The Observer (Version 5.0; Noldus, Wageningen, the Netherlands),

a program specially designed for behavioral observation. The program allows the quantification

of the duration, frequency and serial order of defined therapeutic actions. For example, imagine

the following sequence of actions: A) a physical therapist offers an infant a toy in the midline;

B) after 6 seconds the therapist facilitates rolling behavior with the pelvis or legs as keypoint, C)

meanwhile the therapist gives the caregiver instructions on handling during rolling movements.

In the Observer action A is scored as category H (see Appendix 1, p. 188; the infant is challenged

to produce motor behavior by himself or herself; activity flows over into therapeutic handling).

As the toy is offered in the midline only, the action is performed with little variation (H1). Action

B is scored as category C1 (facilitation techniques - handling), but at the same time the physical

therapist interferes with the infant’s behavior and interrupts his or her activity (category A.4.1).

Action C is scored as category A.4.3 (caregiver training). Start and stop of an action are indicated

by pressing a key on the computer keyboard. It is important to note that The Observer program al-

lows scoring of multiple actions occurring simultaneously (eg, action B). To compare the results of

different treatment sessions, the duration of the PT actions was converted into a relative duration,

which represented a percentage of the total treatment time. Relative duration (%) = (Time spent on

PT action / Total time of treatment session) * 100.

Psychometric quality of the observation protocol

Completeness of the protocol was tested by checking whether all possible PT actions observed

by the researchers were covered by the categories of the observation protocol. Completeness was

considered satisfactory if major part of the PT actions (> 90% of the observation time) could be

classified into protocol categories. Mutual exclusiveness was determined by means of the in-

ter- and intrarater agreement. Substantial agreement indicates that a single PT action is clearly

related to a specific category of the observation protocol. Reliability of the observation protocol

94 95

was measured through inter- and intrarater agreement. Three time intervals, each lasting five

minutes, were selected from within each of 5 randomly selected treatment sessions (n=15). The

intervals 100-400 seconds, 500-800 seconds and 1000-1300 seconds were analyzed. The start of

the treatment session, that is, the first 100 seconds, was excluded from the analyses because dur-

ing this period mainly preparatory actions were performed. Interrater agreement was assessed

by comparing observational scores of the third author with those of the first author. Intrarater

agreement was assessed by comparing the observational scores of the first author who reanalyzed

the video sequences with an interval of at least three weeks.

Data Analysis

The data were analyzed using the computer package SPSS (version 14.0; SPSS Inc, Chicago, Illinois).

Inter- and intrarater agreements were calculated by intraclass correlations (ICCs) of frequency and

relative duration of PT actions. ICC values between 0.50 to 0.75 were considered to indicate satis-

factory reliability, ICC values exceeding 0.75 indicate good reliability (Portney and Watkins 2000).

For the comparison of the relative duration of the main categories and subcategories of PT ac-

tions and the amount of postural support at 4 months with the data at 6 months, Wilcoxon signed

rank tests were used.

To get an impression of the current state of the daily practice in pediatric physical therapy

in The Netherlands we analyzed whether sessions were dominated by PT actions that are in line

with the concept of NDT in baby treatment13 or by PT actions belonging to the functional ap-

proach. A treatment session was classified as NDT-like when the time spent on the category facili-

tation techniques exceeded the 75th percentile of current group data. Similarly, sessions in which

time spent on the category ‘challenging the infant to produce motor behavior by himself or herself

- action continued by the infant’ exceeded the 75th percentile were classified as functional sessions.

Correlations and differences with a p-value < 0.05 were considered as statistically significant.

results

Observation protocol

The observation protocol contains three levels of observation. The first level consists of eight

mutually exclusive main categories of PT actions (see Appendix 1). The second level consists of

the subcategories of the main techniques. For example, the main category facilitation techniques

has the following subcategories: handling, pressure techniques, transitions and support devices.

The third level of the protocol entails concrete PT-actions. For example, in the supine or sitting

position: shoulders function as a key point, handling hands guide the shoulders of the infant in

protraction to control the infants movements and to facilitate hand-hand contact and symmetry.

The degree of postural support provided by the physical therapist or caregiver when the infant

was in prone, side or sitting position was scored as an additional variable. Postural support was

classified into four categories: no postural support, minimal postural support, clear postural sup-

port and full postural support

Psychometric properties

The contents of 42 physical therapy sessions were analyzed. For two infants, only one video

recording was available for observation. In one case, the therapist had already discontinued treat-

ment before 6 months. In the other case, the family was on holiday when the video recording was

scheduled.

Major part of the observed PT actions could be classified into the categories of the observa-

tion protocol. Only 3% of the duration of physical therapy sessions could not be classified into the

categories of the observation protocol, that is, they were classified in the category ‘not specified’.

This indicates that the observation protocol meets the criterion for completeness.

Inter- and intrarater agreement on the frequency of PT actions was moderate to good with

ICCs varying from 0.68 to 1.00 (interrater) and 0.70 to 0.98 (intrarater). The same was true for the

inter- and intrarater agreement on the relative duration of actions; ICCs ranged from 0.76 to 1.00

(interrater) and 0.69 to 0.99 (intrarater; Table 5.2). The confidence intervals of the PT actions are

presented in Table 5.2. The data indicate that the PT actions of the observation protocol can be

assessed in a reliable way. Moreover the moderate to good inter- and intrarater agreement points

to a sufficient degree of mutually exclusiveness.

Relative duration of therapeutic actions

The data on frequency and relative duration of PT actions were highly correlated (Spearman

rank correlation at 4 months: 0.89, at 6 months: 0.88). This suggests that both parameters carry

virtually identical information. We therefore decided to restrict our report on the contents of the

sessions to one parameter. We chose relative duration as it had slightly better inter- and intrarater

agreement values than frequency.

The relative duration of the majority of the PT actions applied at 4 months did not differ sig-

nificantly from those at 6 months (Table 5.3). The only exceptions were challenging the infant to

produce motor behavior by himself which flows over into handling, which occurred slightly more

at 6 months than at 4 months (p=0.03) and in the provision of postural support. Less postural sup-

port was provided during PT actions in infants aged 6 months than in those aged 4 months (Table

5.3).

In the following report on the contents of the NDT sessions, we pooled the 4- and 6-month

data for those actions in which the relative duration of PT actions at 4 and 6 months were identi-

cal. Table 5.3 shows that the application of physical therapy in infancy is characterized by a large

heterogeneity. Most of treatment time was spent with PT actions in the categories of motor

behavior produced by the infant, that is, motor behavior that was initiated by the infant and

behavior that was challenged by caregiver or physical therapist who a) let the infant try on her or

his own or b) took over control of the infant’s movements by means of handling (together ~40%),

and facilitation techniques (29%; Figure 5.1). We noted that during both challenging the infant to

produce motor behavior by himself/herself, which flows over into handling (5.6%) and challenging

the infant to produce motor behavior by himself/herself which is continued by the infant (13.2%),

96 97

tab

le 5

.2. I

nter

rate

r an

d in

trar

ater

agr

eem

ent (

ICC

) on

mai

n an

d su

bcat

egor

ies

of p

hysi

othe

rape

utic

ac

tion

s (f

requ

ency

and

rel

ativ

e du

rati

on o

f PT

act

ion

s; n

=15

vide

o fr

agm

ents

of 5

min

utes

).

Inte

rven

tion

In

terr

ater

agr

eem

ent

Intr

arat

er a

gree

men

tFr

eque

ncy

(n=1

5)D

urat

ion

(n=1

5)Fr

eque

ncy

(n=1

5)D

urat

ion

(n=1

5)

ICC

(95%

CI)

ICC

(95%

CI)

ICC

(95%

CI)

ICC

(95%

CI)

Fam

ily

invo

lvem

ent a

nd E

duca

tion

al c

ompo

nent

E

duca

tion

al a

ctio

ns

0.78

(0.4

8-0.

92)

0.78

(0.4

8-0.

92) b

0.80

(0.4

9-0.

93) b

0.88

(0.6

6-0.

96) b

- Car

egiv

er in

terf

eres

wit

h in

fant

’s ac

tivi

ties

n.o

.an

.o.a

n.o

.an

.o.a

- PT

inte

rfer

es w

ith

infa

nt’s

acti

viti

es

0.76

(0.4

3-0.

91)

0.76

(0.4

3-0.

91) b

0.89

(0.7

0-0

.96)

b0.

80 (0

.51-

0.93

) b

- PT

gui

des

the

infa

nt1.

00 (1

.00

-1.0

0)0.

99 (0

.98-

1.00

) b0.

74 (0

.38-

0.90

) b0.

75 (0

.40

-0.9

0) b

- PT

giv

es c

areg

iver

tra

inin

gn

.o.a

n.o

a0.

95 (0

.86-

0.98

) b0.

78 (0

.46-

0.92

) b

- PT

coa

ches

the

car

egiv

ern

.o.a

n.o

.an

.o.a

n.o

.a

Com

mun

icat

ion

0.87

(0.6

6-0.

95)

0.98

(0.9

4-0.

99) b

0.89

(0.6

9-0.

96) b

0.96

(0.8

0-0

.98)

b

- In

form

atio

n ex

chan

ge0.

87 (0

.68-

0.95

)0.

98 (0

.93-

0.99

) bn

.o.a

n.o

.a

- C

onte

nts

of in

form

atio

n0.

81 (0

.54-

0.93

)0.

83 (0

.59-

0.94

) b0.

86 (0

.63-

0.95

) b0.

81 (0

.52-

0.93

) b

- In

stru

ct0.

86 (0

.64-

0.95

)0.

81 (0

.54-

0.93

) b0.

85 (0

.61-

0.95

) b0.

83 (0

.57-

0.94

) b

- P

rovi

de fe

edba

ck0.

78 (0

.48-

0.92

)0.

96 (0

.89-

0.99

) b0.

82 (0

.54-

0.94

) b0.

87 (0

.65-

0.95

) b

- Im

part

kno

wle

dge

0.87

(0.6

8-0.

95)

0.91

(0.7

6-0.

97) b

0.75

(0.4

7-0.

92) b

0.74

(0.3

9-0.

91) b

Neu

rom

otor

com

pone

ntFa

cilit

atio

n te

chn

ique

s

0.95

(0.8

6-0.

98)

0.97

(0.9

0-0

.99)

b0.

97 (0

.91-

0.99

) b0.

99 (0

.99-

1.00

) b

- Han

dlin

g 0.

72 (0

.37-

0.89

)0.

80 (0

.52-

0.93

) b0.

95 (0

.86-

0.98

) b0.

91 (0

.76-

0.97

) b

- Pre

ssur

e te

chn

ique

s0.

97 (0

.91-

0.99

)0.

88 (0

.69-

0.96

) b0.

93 (0

.80

-0.9

8) b

0.97

(0.9

2-0.

99) b

- Sup

port

dev

ice

n.o

.an

.o.a

n.o

.an

.o.a

- Tra

nsi

tion

0.86

(0.6

4-0.

95)

0.91

(0.7

5-0.

98) b

0.89

(0.6

9-0.

96) b

0.87

(0.6

5-0.

95) b

Sen

sory

exp

erie

nce

0.85

(0.6

3-0.

95)

0.88

(0.7

0-0

.96)

b0.

76 (0

.42-

0.91

) b0.

78 (0

.46-

0.92

) b

Pass

ive

mot

or e

xper

ienc

e0.

71 (0

.35-

0.89

)0.

91 (0

.77-

0.97

) b0.

70 (0

.70

-0.8

9) b

0.92

(0.7

9-0.

97) b

Self

-pro

duce

d m

otor

beh

avio

r –

no in

terf

eren

ce0.

95 (0

.85-

0.98

)0.

97 (0

.93-

0.99

) b0.

82 (0

.53-

0.93

) b0.

94 (0

.83-

0.98

) b

Cha

lleng

ed to

sel

f-pr

oduc

ed m

otor

beh

avio

r –

acti

on c

onti

nued

by

infa

nt0.

86 (0

.64-

0.95

)0.

80 (0

.51-

0.92

) b0.

86 (0

.63-

0.95

) b0.

86 (0

.62-

0.95

) b

- Lit

tle

vari

atio

n0.

90 (0

.74-

0.96

)0.

87 (0

.67-

0.95

) b0.

85 (0

.60

-0.9

5) b

0.83

(0.5

7-0.

94) b

- Lar

ge v

aria

tion

0.75

(0.4

1-0.

90)

0.82

(0.5

5-0.

93) b

0.84

(0.5

9-0.

94) b

0.89

(0.7

1-0.

96) b

Cha

lleng

ed to

sel

f-pr

oduc

ed m

otor

beh

avio

r –

wit

h ov

erflo

w in

to h

andl

ing

0.80

(0.5

2-0.

93)

0.88

(0.7

0-0

.96)

b0.

84 (0

.59-

0.94

) b0.

72 (0

.34-

0.90

) b

- Lit

tle

vari

atio

n0.

85 (0

.62-

0.95

)0.

84 (0

.59-

0.94

) b0.

86 (0

.64-

0.95

) b0.

72 (0

.35-

0.90

) b

- Lar

ge v

aria

tion

0.72

(0.3

6-0.

89)

0.90

(0.7

4-0.

96) b

n.o

.an

.o.a

Not

spe

cifi

ed a

ctio

ns

0.68

(0.3

0-0

.88)

0.84

(0.6

1-0.

94) b

0.81

(0.5

2-0.

93) b

0.79

(0.4

9-0.

93) b

No

post

ural

sup

port

0.93

(0.8

1-0.

98)

0.99

(0.9

7-1.

00)

0.97

(0.9

1-0.

99)

0.98

(0.9

5-0.

99)

Min

imal

pos

tura

l sup

port

0.78

(0.4

8-0.

92)

0.89

(0.7

2-0.

96)

0.77

(0.4

4-0.

92)

0.69

(0.2

9-0.

88)

Cle

ar p

ostu

ral s

uppo

rt0.

69 (0

.31-

0.88

)0.

89 (0

.70

-0.9

6)0.

89 (0

.71-

0.96

)0.

89 (0

.70

-0.9

6)Fu

ll po

stur

al s

uppo

rt0.

69 (0

.31-

0.88

)1.

00 (0

.99-

1.00

)0.

98 (0

.94-

0.99

)0.

91 (0

.74-

0.97

)

95%

CI =

95%

con

fiden

ce in

terv

al, I

CC

= in

trac

lass

cor

rela

tion

coe

ffici

ent

a Not

obs

erve

d b p

<.05

; for

defi

nit

ion

of t

he v

ario

us c

ateg

orie

s se

e A

ppen

dix

98 99

infants were challenged to explore only one movement strategy. The most frequently applied

subcategory of facilitation techniques was handling. About 9% of treatment time was spent on

sensory and passive motor experience. At 6 months, less postural support was provided during PT

actions (Table 5.3).

The data indicated that the 75th percentile of the category facilitation techniques matched 44%

of treatment time, whereas the 75th percentile of the category challenging the infant to produce

motor behavior by himself/herself which is continued by the infant was at 20% of treatment time.

In ten sessions, more than 44% of the treatment time was spent of facilitation techniques indica-

ting that these sessions could be classified as NDT-like. Another 10 sessions could be classified

as functional as more than 20% of treatment time was spent with the category challenging the

infant to produce motor behavior by himself/herself, which is continued by the infant, leaving 22

sessions with mixed contents.

During all treatment sessions, one of the caregivers was present and observed what the physical

therapist was doing. In 12 sessions caregivers acted together with the physical therapist to guide the

attention of the infant. Approximately 4% of the time was devoted to family involvement and edu-

cational actions. This usually consisted of educational actions for example interfering with infant’s

activities. Very little treatment time was devoted to the training of caregivers on how to continue

treatment strategies during daily life activities and/or in the home environment. Treatment related

communication between physical therapist and caregiver was more or less equally distributed

between information exchange, imparting knowledge, giving feedback, and sharing information

regarding handling and the application of intervention strategies into daily routines.

Discussion and Conclusion

The present study demonstrated that it is possible to assess PT actions during physical therapy of

young infants at high risk for developmental disorders in a systematic, standardized, and reliable

way. Before we address physiotherapeutic considerations, we discuss the strengths and weak-

nesses of our study.

One of the limitations of the study is the sample size, which results, for example, in relatively large

confidence intervals for the ICC values of intra- and interrater agreement. Confidence intervals

were especially large in infrequently occurring PT actions. The large confidence intervals indicate

that the results cannot be generalized to any population. Therefore, it is important to realize that

the results of this study should be interpreted with caution.

Another problem is that at young age CP cannot be diagnosed. This means that physical

therapy at very early age is applied to infants at risk for developmental disorders, including CP, not

to infants with CP. This study group was selected on the basis of definitely abnormal GMs around

the age of 3 months corrected age. The presence of definitely abnormal GMs at this age is a power-

ful indicator of developmental disorders, including CP (Prechtl et al 1997, Prechtl 2001, Hadders-

Algra 2004). Most of the infants participating in the study continued to show neurological

dysfunction during the intervention period indicating they had need of ongoing physical therapy.

Cat

egor

ies

of P

t a

ctio

ns

relative duraction (% of treatment time)

0 10

28,6%

6,1%

2,4%

21,4%

5,5%

13,2%

3,9%

12,3%

3,2%

16,2%

3,5%

19,3%

5,1%

20 30 40 50 60 70 80 90 100

Facilitation-Inhibition

Sensory experience

Passive experience

SPMB no interference

CIPMB --> handling

CIPMB continued by infant

Family and education

Communication

Not specified

no postural support

minimal postural support

clear postural support

full postural support

The major strength of this study is that we succeeded in developing a standardized observation

protocol that permitted analysis of the black box of physical therapy interventions for infants. The

protocol is an instrument that may be used to assess heterogeneity in physical therapy as it offers

a tool to describe operationally what therapists do in a treatment session, both across varying

frames of theoretical references and across countries. The video analysis of the contents of physi-

cal therapy sessions is relatively time-consuming because it is performed on a real-time basis and

usually multiple runs are needed to track down simultaneously occurring actions. Experience at

our institute indicates that the technique can be learned relatively quickly and reliably. The analy-

sis of PT actions by means of the standardized observation protocol paves the way for an objective

interpretation of the contents of various pediatric physical therapy sessions.

Physiotherapeutic considerations and implications for future research

It was possible to develop an observation protocol to assess the contents of physical therapy for

young infants. Eight main categories were developed that allowed the classification of single PT

actions. During the developmental process, the observation protocol was checked for psychomet-

ric properties, such as completeness, mutual exclusiveness, and reliability. The results showed that

the observation protocol caught virtually all PT actions during the treatment of young infants

CIPMB = Challenging Infant to Produce Motor Behavior, SPMB = Self Produced Motor Behavior

Figure 5.1. Relative duration (% of treatment time) of PT actions and amount of postural support (range and mean value). Results of pooling of 4 and 6 months data.

100 101

Intervention 4 mo 6 mo Pooled data 4 and 6 mo

Med

ian

ran

ge (%

)

Med

ian

ran

ge (%

)

p Med

ian

ran

ge (%

)

Family involvement and Educational component

Educational actions 4.1 1-37 3.8 1-22 0.94 4.0 1-37- Caregiver interferes with infant’s activities n.o.a n.o.a

- PT interferes with infant’s activities 2.1 0-32 2.0 0-17 0.30 2.1 0-32- PT guides the infant 0 0- 1 0.2 0-10 0.88 0.2 0-10- PT gives caregiver training 0.1 0-13 0 0- 5 0.65 0 0-13- PT coaches the caregiver n.o.a n.o.a

Communication 10.5 0-55 15.6 0-45 0.99 12.4 0-55 - Information exchange 0.7 0-20 1.4 0-31 0.44 1.3 0-31 - Contents of information 1.0 0-16 1.4 0-14 1.5 0-15 - Instruct 0.3 0- 4 0.2 0- 4 0.3 0-4 - Provide feedback 3.5 0-17 3.1 0-11 0.85 3.3 0-17 - Impart knowledge 2.3 0-17 1.4 0-10 0.21 1.7 0-17

Neuromotor componentFacilitation techniques 31.7 5-74 28.1 7-54 0.28 28.6 5-74

- Handling 16 4-57 16.5 3-42 0.63 16.3 3-57- Pressure techniques 6.3 0-25 5.3 0-24 0.25 6.2 0-25- Support device 0 0-25 0 0-15 0.27 0 0-25- Transition 4.7 0-17 4.0 1-16 0.96 3.8 0-17

Sensory experience 8.5 1-21 6.1 0-36 0.64 6.1 0-36Passive motor experience 4.6 0-20 2.0 0-19 0.09 2.4 0-20Self-produced motor behavior – no interference

19.5 0-73 21.3 1-83 0.10 21.3 0-81

Challenged to self-produced motor behavior – action continued by infant

15.9 0-32 12.3 2-38 0.20 13.2 0-38

- Little variation 12.4 0-28 8.3 1-30 0.11 9.1 0-30- Large variation 1.3 0-16 0 0-27 0.45 1.0 0-27

Challenged to self-produced motor behavior – with overflow into handling

5.5 1-25 5.6 1-37 0.03*

- Little variation 4.9 0-23 3.7 0-37 0.04*- Large variation 0 0-25 0 0- 6 0.33 0 0-25

Not specified actions 1.9 0-13 3.5 0-11 0.16 3.2 0-13

No postural support 8.2 0-41 20.2 3-45 0.03*Minimal postural support 2.1 0-11 5.0 1-34 0.05*Clear postural support 13.9 2-36 22.1 1-42 0.01*Full postural support 8.2 0-66 4.2 0-32 0.20 5.1 0-66

table 5.3. Mean relative duration of PT actions and amount of postural support at 4 and 6 months corrected age (% of time) and results of Wilcoxon signed-rank tests. Right hand column: pooling of 4 and 6 months data.

a Not observed * Wilcoxon signed-rank test: p<.05; for definition of the various categories see Appendix

and that it had a good inter- and intrarater reliability. The good reliability also indicated that the

categories in the observation protocol were mutually exclusive. The small proportion of treatment

time (3%) which could not be classified into the categories of the observation protocol mainly

was spent on comforting the infant and physical therapist or caregiver and infant being out of

camera view. We may thus conclude that our observation protocol is an appropriate instrument

for the opening of the black box of physical therapy for infants with a high risk for developmental

disorders.

The data represent the contents of various pediatric physical therapy sessions. They give an

indication of how physical therapy is applied to young infants at high risk for developmental

disorders in the Netherlands. The data showed that the relative duration of the applied treatment

techniques covers a wide range, implying a large heterogeneity in the practical implementation of

physical therapy for infants. Presumably, this does reflect reality because part of the PT actions

that are performed belong to the concept of NDT and baby treatment, which by itself are well-

known for its heterogeneity in application (Bly 1991). The combination with PT actions from a

more functional point of view, such as the encouragement of self produced motor behavior, makes

treatment application even more diverse. Presumably, the heterogeneity is brought about by

the evolution of treatment techniques and theoretical assumptions over the years. First, physi-

cal therapists became aware that motor achievements occurring after the application of former

treatment techniques, such as those of NDT, did not automatically carry over into activities of

daily life. Nowadays, goals are defined more in relation to function, the needs of the caregiver, and

new treatment approaches have been incorporated into older ones (Howle 2002). With respect to

our ‘Opening of the Black Box’ tool, it is important to note that – despite the heterogeneity in the

implementation of physical therapy for young infants – virtually all PT actions could be classified

with the help of the protocol.

Across the world, treatment application in infancy varies from NDT to Vojta and from devel-

opmental interventions to stimulation programs. Thus, it would be interesting to know whether

the protocol can be used also to evaluate physical therapy for infants in other countries. This

study indicates that also within the application of a specific approach in one country heterogene-

ity is present.

Physical therapists frequently expressed surprise when they got feedback on the contents of

their treatment sessions. They became aware of the discrepancy between what they believed that

they had been doing and what they actually had been doing. This finding illustrates the difficulty

of verbal communication. Success of verbal communication depends not only on the accuracy of

the speaker or author and the intention of the hearer or reader to interpret as precisely as possible

but also on the body of knowledge and the verbal frame of reference present in sender and re-

ceiver. These frames of reference may differ more often than we realize, resulting in miscommuni-

cation. Our findings suggest that the application of video feedback may serve as an instrument to

prevent this type of misunderstanding. Video recording thus may be a useful tool in the training

of novice pediatric physical therapists.

102 103

This study indicates that the categories challenging the infant to produce motor behavior by

himself and facilitation techniques were applied most. Three types of motor behavior produced

by the infant were distinguished: behavior which was initiated by the infant and behavior which

was challenged by caregiver or therapist, who a) let the infant try on her/his own or b) took over

control of the infant’s movements by means of handling. We observed that self initiated activity of

the infant usually occurred when the therapist started to communicate with the parents, leaving

the infant in a situation in which his or her capacities were not challenged. Another observation

was that during motor skill practice the physical therapist usually challenged the infant to explore

only one movement strategy. This means that during the physical therapy sessions of this study,

little variation in motor behavior was practiced. This is surprising because current concepts of in-

fant motor dysfunction suggest that varied practice is beneficial for motor development (Hadders-

Algra 2000b).

The protocol pays specific attention to family involvement and educational actions. In the

latter, we distinguished between educational actions regarding the child and educational actions

regarding the caregiver, that is, caregiver training and caregiver coaching. It was remarkable that

during treatment, little time was spent training the caregiver on how to apply treatment strate-

gies during daily life activities and/or in the home environment. According to the traditional

treatment approach, merely based on NDT, guiding and training of caregivers are the primary

technique to achieve carryover from treatment to everyday life (Bobath and Bobath 1984, Finnie

1996, Howle 2002). In the functional approaches caregivers are the key persons in the child’s

development and education (Law et al 1998, Rosenbaum et al 1998, Ketelaar et al 2001, Ekström et

al 2005). Thus, it seems that actual practice of the therapists involved in this study differed from

current notions because caregivers mainly acted as a passive observer of treatment instead of be-

ing actively involved and instructed.

Future research should aim firstly at a replication of this study in infants at various ages, with

various degrees of neurological dysfunction, and in various countries. A next step will be to relate

the contents of infant physical therapy sessions to developmental outcome. This knowledge might

shed light on the potentially effective and ineffective elements in infant physical therapy.

In conclusion, this study demonstrated that it is possible to assess PT actions during physical

therapy of young infants at high risk for CP in a systematic, standardized, and reliable way despite

the heterogeneity in the implementation of infant physical therapy. The study indicated that

opening the black box of infant physical therapy has shown that in the Netherlands the applica-

tion is very heterogeneous and varies between more traditional (NDT-like) to more functionally

oriented treatment. Our study may be seen as a first step in the development of a tool to describe

therapists’ actions during physical therapy interventions. Objective knowledge on the contents of

physical therapy sessions is an essential step towards evidence-based practice. The next steps to be

taken are replication studies on reliability assessment, studies in different populations (eg, older

children with CP or developmental coordination disorder) and in different countries, and studies

connecting the contents of physical therapy sessions with developmental outcome.

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C H a P t E r 6

dI f f e r e nc e s be t w e e n

t h e fA m I ly-c e n t e r e d

P ro g r A m “c oP I ng A n d

c A r I ng f or I n fA n t s

w I t h s P e c I A l n e e d s”

A n d I n fA n t t r e At m e n t

b A s e d on P r I nc I P l e s

of u rode V e l oPm e n tA l

t r e At m e n t --------------------------------------------------------------------------------------------------------------

Tineke Dirks, Cornill H. Blauw-Hospers, Lily J. Hulshof, and Mijna Hadders-Algra

Submitted for publication

106 107

abstract

Background. Evidence for effectiveness of pediatric physical therapy in infants at high risk for

developmental motor disorders is limited. Therefore, we have developed the early intervention

program COPCA: COPing with and CAring for infants with special needs – a family-centered

program. COPCA is based on two components: 1) family involvement and educational parenting,

2) the neuromotor principles of the Neuronal Group Selection Theory. The COPCA coach uses

principles of coaching to encourage the family’s own capacities for solving problems of daily care

and incorporate variation, along with trial and error in daily activities.

Objective. To evaluate whether the content of COPCA sessions differs from that of traditional

infant physical (TIP) therapy sessions.

Design. Quantitative video analysis of therapy sessions with infants participating in a randomized

controlled trial.

Subjects and methods. Forty-six high-risk infants were randomly assigned to receive COPCA

(n=21) or TIP (n=25) between 3 and 6 months corrected age (CA). TIP in the Netherlands is gene-

rally based on NeuroDevelopmental Treatment (NDT) principles. Intervention sessions were

videotaped at 4 and 6 months CA, and analyzed with a standardized observation protocol for the

classification of physiotherapeutic (PT) actions. Outcome parameters were relative amounts of

time spent on specific PT actions.

results. The content of COPCA and TIP differed substantially. For instance, in TIP sessions 20

more time was spent on facilitation techniques, including handling (29% vs. 3%, p<0.001) than in

COPCA sessions. During COPCA more time was spent on family coaching and education (16% vs.

4%, p<0.001) than during TIP.

Conclusion. COPCA differs broadly from TIP in terms of its NDT basis, justifying the label “novel

program.” Studies on the effectiveness of this family-centered program are required.

Introduction

Worldwide, therapists are trained in specialized infant treatment based on the principles of the

NeuroDevelopmental treatment (NDT) approach. The original philosophy – the basic assump-

tions and clinical concepts of NDT in infancy – can be attributed to Karl and Bertha Bobath

(Bobath 1967) and Elsbeth Köng (Köng 1966), whereas Mary Quintin was responsible for a major

part of the practical implementation of the Bobath principles in infant treatment (Bly 1999).

However, daily practice of the approach in different countries and healthcare settings reveals that,

since then, numerous modifications and changes in the implementation of infant treatment have

occurred.

Notwithstanding the occurrence of substantial changes in NDT treatment in infancy, three

recent systematic reviews on the effect of early intervention in infants at high risk for develop-

mental disorders indicated that application of NDT does not result in improved developmental

outcome (Blauw-Hospers and Hadders-Algra 2005, Blauw-Hospers et al 2007, Spittle et al 2007).

The reviews also indicated that best results are achieved by application of specific motor-training

programs and general developmental programs.

These findings and novel insights into the biological and psychological principles governing

motor development after a lesion of the brain at early age (Hadders-Algra 2000b, Hadders-Algra

2008b, Diamond 2009), along with an evolving shift in the manner in which family-centered

services (FCS) are delivered, have inspired us to develop the COPCA program (Dirks and Hadders-

Algra 2003). During the last few decades it has become clear that “family-centered” is a crucial

aspect of interventions applied in infants and young children (Als 1992, Rosenbaum et al 1992,

Dunst and Trivette 1996, McBride and Petersen 1997, Law et al 1998, King et al 2004, Rosenbaum

2004, Sanders et al 2004, Nijhuis et al 2007).

COPCA, that is, COPing with and CAring for infants with special needs, a family-centered

program, is an intervention program for families of infants at high risk for developmental motor

disorders after term age. The theoretical contents of COPCA differ broadly from Traditional

Infant Physical Therapy (TIP) based on NDT (see Table 6.1 for an overview). Before we move on to

the actual study, we should first summarize the principles of the COPCA program and TIP based

on NDT.

COPCa

COPCA is rooted in the framework of the International Classification of Functioning, Disabil-

ity and Health for Children and Youth (World Health Organisation 2007). It aims to promote

activities and participation of the family, including the infant with special needs, while taking

into account the limitations imposed by the infant’s bodily impairments. COPCA consists of two

components:

1. A family involvement and educational parenting component in which the key elements are: a)

family autonomy in decision-making processes and family responsibility for the quality of life

(Law et al 1998) and b) family-specific education, that is, the family-specific styles of parents for

108 109

educating the child to independency and personal responsibility. This means that COPCA not

only respects the autonomy of the family in the process of parenting, but also values the role of

the individual characteristics of both caregiver and child within the process (Scarr 2001, Sanders

et al 2004).

2. A neurodevelopmental component based on the principles of the Neuronal Group Selection

Theory (Edelman 1989).

The major aims of COPCA, which are listed below, are closely related to these two components.

1. The family involvement and educational component

A fundamental idea of COPCA is the transactional perspective on patterns of reciprocal contin-

gent interactions between caregiver, siblings, and infant, along with the notion that changing pat-

terns of behavior are a characteristic of early development (Fiese 1997, De Weerth and Van Geert

2002). This means that the family is regarded as a dynamic system of bidirectional dyads. The

family itself is nested in a larger system including societal influences, socioeconomic factors, and

current living conditions (Bronfenbrenner 1967). COPCA aims to encourage family responsive-

ness, in other words, it aims to promote the capacity of those involved in the family relationships

in order to respond appropriately to each other’s signals, communication, and changing states.

In the case of infants with special needs, responsiveness includes the caregiver’s recognition of

the signals of the infant and the caregiver’s responses to the actual needs of the infant, and not

to preconceived ideas of what the baby might need (Mahoney 1998). To achieve these goals, the

principles of coaching are used. The term “coaching” has various connotations. For instance, Als

(1992) uses verbal and non-verbal coaching strategies to instruct parents in the neonatal intensive

care unit on how to observe and analyze the infant’s neurobehavior and to hold, handle, feed, and

bathe the infant. In NDT, treatment coaching is used to teach parents special therapeutic skills

such as handling. NDT provides parents with references about what the therapist is doing and

furnishes parental assignments while the therapist treats the infant (Howle 2002). The therapist

uses parent training to teach parents what they have to do or could do in daily care activities.

In COPCA, coaching implies the promotion of creative exploration of the competencies of the

family members, including the infant with special needs, in order to stimulate self-made decisions

and improve the quality of life (Hamlin et al 2009). In other words, COPCA encourages families to

follow their own ideas. This is a strategy that fits the following parental expression. “Don’t tell me

what I can do, have to do, or must do; help me to discover it by myself.” This also implies that the

coach does not have an instructional role but instead supports family members on the basis of an

ongoing equal partnership in order to reveal their competencies, goals, desires, and hopes. To this

end, the coach uses focused bidirectional, nuanced, and detailed conversations. An important as-

pect of the process of coaching is the unconditional acceptance of the responsibility of the family.

COPCA coaches the family by creating a process in which the family’s needs and wants are

translated into solutions for how to cope with the problems related to the infant’s development,

while respecting the roles, culture, traditions, and routines of the family (Fiese 1997), along with

the family’s own educational perspective. The coach listens, informs, and observes while the

caregiver is involved in daily routines with the child, including play, thereby creating a situation

in which the caregivers feel free to explore and discuss alternative strategies. Specific attention is

paid to the role of siblings in care and play, along with sibling well-being. The above implies that

COPCA is not a program in which professionals, parents, or others are engaged in “treating” the

infant. COPCA is a family-centered program for professionals involved in the care of infants with

special needs such as physical therapists. The aim of the program is to encourage the family’s

own capacities for solving the problems of daily care in naturally occurring parenting situations.

Therefore, the program is delivered preferably in the home environment. Our primary target as

far as professionals are concerned is pediatric physical therapists, since infants at high risk for

motor disorders are generally referred to these professionals. However, other professionals, such

as nurses, special educators, social workers, or occupational therapists could also become COPCA

coaches. To underline the importance of coaching, we would rather call the professional applying

the COPCA program a “coach” rather than a “therapist.”

The problems of parenting are often complex and may vary from concerns about the infant’s

future locomotor development, feeding difficulties, and choice of toys, to concerns about under-

standing infant behavior and educational problems such as to what extent should a parent inter-

fere with the infant’s actions or whether siblings should be allowed to play with the infant. Other

factors which may contribute to family distress are social issues such as time management (how

to distribute time between child, siblings, partner, family, friends, work, etc.) and concerns about

service delivery.

COPCA acknowledges that raising a child is a life-long challenge. Raising an infant with

special needs due to an early lesion of the brain and/or neurological dysfunction adds to this

challenge (Brehaut et al 2004, Raina et al 2005). Some examples of additional challenges which

caregivers of infants with special needs may encounter and the way COPCA coaches deal with

them are:

• The uncertain developmental of an infant with neurological dysfunction and/or an early lesion

of the brain may induce parental stress (Rentinck et al 2007). Coaching addresses these uncer-

tainties about the child’s health, prognosis, and need for special services. The family members

are encouraged to improve their personal coping skills.

• Stress may be associated with processes of bereavement – the mourning associated with the

loss of the prospect of a typically developing child – when the chances of persisting disability

are high (Kearney and Griffin 2001). Typically the process of mourning is a step-by-step process,

involving cycles of feelings of disappointment, sadness, grief, anger, and guilt, which occur each

time that caregivers realize that the future prospects have to be adjusted once again. COPCA

focuses on discussing these processes with family members and encouraging the family to accept

and express feelings of bereavement. COPCA coaches highlight the family’s own strengths and

capacities, and discuss the possibility of external sources of support such as relatives, neighbor-

hood, friends, or the religious community, or the option of professional help.

110 111

• The infant’s neurological dysfunction may induce atypical infant behavior which may inter-

fere with intuitive parenting capacities (Papousek and Papousek 1984). COPCA focuses on the

promotion and restoration of the intuitive parenting capacities by explaining to caregivers the

mechanisms of caregiver-infant interaction and the effect of neurological dysfunction on this

interaction (Sameroff and McKenzie 2003). It also stresses that parenting is a developmental

process involving trial and error, in addition to life-long learning on the part of both caregiver

and child (Cowan 1998). Specific attention is paid to educational actions, including play in child-

preferred activities whereby the motor principles of NGST are taken into account.

• The infant’s neurological dysfunction and/or an early lesion of the brain may result in atypical

motor development. This may introduce the need for additional equipment such as a special

chair or a supporting device for walking. COPCA coaches inform and discuss with caregivers the

possible strategies and options for handling these additional needs, and it is here that the motor

principles of NGST are taken into account.

• The prospect of having a child with a persisting disability introduces many practical and emo-

tional problems into the family, problems which may vary from practical care issues for the child

with special needs to dealing with service providers and organizing technical adaptations to the

living environment (Raina et al 2005). COPCA focuses on coaching the family members in such

a way that they are fully able to participate in the community, including the practical problems

related to the presence of an infant with special needs.

In general, pediatric physical therapists are trained in addressing issues of motor development

and motor dysfunction, while at the same time providing their work within the framework of

family-centered philosophy (Bly 1999, Chiarello and Effgen 2006). Nevertheless, translation of the

principles of family-centered services into the actual behavior of service providers in an appropri-

ate family-centered approach may be difficult (O’Neill and Palisano 2000, Nijhuis et al 2007). This

is illustrated by the findings of O’Neil and coworkers (2000) which suggest that therapists who be-

lieve in family-centered care consider the aspects of functional activities and child characteristics

– reflective of a child-focused perspective – as being of paramount importance in clinical decision

making. COPCA’s perspective on family-centered care is to empower the family to use their own

competences and problem-solving skills in the process of coping with the daily task of caring for

and rearing a child with special needs. This implies a shift from family-centered care with a child

focused perspective to a family-relationship-focused perspective in which the motor principles of

NGST are only one of the elements taken into account

COPCA recognizes that professionals may perceive limitations in the coaching approach, since

family autonomy implies limitations in terms of the extent to which educational action problems

and difficulties in the community may be resolved. In those cases where the infant has complex

morbidity, for instance, motor dysfunction associated with serious feeding, speech, and language

problems, low vision, or in those cases where a need for additional equipment arises, the COPCA

coach functions as one of the members of the interdisciplinary team involved in guiding the family.

2. The Neurodevelopmental component: NGST

According to NGST, typical motor development is characterized by two phases of variability: the

phases of primary and secondary variability. The borders of variability are determined by genetic

instructions (Edelman 1989, Hadders-Algra 2000a, 2008a). During the phase of primary vari-

ability, motor behavior is characterized by abundant variation. The motor variations represent a

fundamental developmental phenomenon; they are brought about by explorative activity of the

nervous system. The exploration generates a wealth of self-produced afferent information which,

in turn, is used for further shaping of the nervous system. At that stage, the afferent information

is not used for adaptation of motor behavior to environmental constraints. In other words, the

phase of primary variability is characterized by variable but non-adaptive motor behavior.

At a certain point in time, the nervous system is able to use the afferent information produced

by behavior and experience in order to select the motor behavior which fits the specific situation

best; the phase of secondary or adaptive variability then starts. This selection process is based on

active self-produced sensorimotor trial and error experiences. The transition from primary to

secondary variability occurs at function-specific ages. For instance, in the development of sucking

behavior the phase of secondary variability starts prior to term age, and in the development of foot-

placing during walking it starts between 12 and 18 months (Hadders-Algra 2000a). Around the age

of 18 months all basic motor functions have reached the first stages of secondary variability.

Due to the ingenious interaction between self-produced motor activities with trial and error

learning and the long lasting developmental processes in the brain, it takes until the age of 18

to 20 years before the secondary neural repertoire has obtained its mature, adult configuration

(Hadders-Algra 2008a).

In terms of NGST an early lesion of the brain has two major consequences (Hadders-Algra

2000b, 2008b). First, the repertoire of motor strategies is reduced. This results in less variable

and more stereotyped motor behavior. In addition, infants with a prenatally, perinatally, or early

postnatally acquired lesion or malformation of the brain have problems with the selection of the

most appropriately adapted strategy out of the repertoire. The selection problems have a dual

origin: they are related to the deficits in the processing of sensory information and to the fact that

the best solution may not be available due to repertoire reduction.

The practical consequences of the above-mentioned problems are twofold. First, the limited

motor repertoire may result in the absence of a specific motor strategy which would be available

as the best solution in a specific situation for a typically developing infant. Due to absence of the

“best” solution, the infant with an early lesion of the brain may have to choose a motor solution

which differs from that of the typically developing infant. This implies that the different motor

behavior of an infant with an early lesion of the brain should not a priori be regarded as deviant,

that is, as something which deserves to be “treated away,” as it may be the infant’s best and most

adaptive solution for the situation (Latash and Anson 1996). Second, due to the deficits in the

processing of sensory information which hamper the process of selection of the best strategy,

infants with an early lesion of the brain need ten- to a hundredfold more active motor experience

112 113

than typically developing children (Valvano and Newell 1998).

Translating NGST principles into daily practice of the COPCA program means that the

COPCA coach will inform and coach family members while taking into account the family’s

educational perspective. Family members receive suggestions for incorporating variation and trial

and error in daily activities, since this might enhance the infant’s motor repertoire and promote

the ability to select the best strategy for different conditions. This also implies that COPCA is

characterized by being “hands-off.” Some examples of variation in activities are those variations in

which the infant is challenged to produce self-initiated motor behavior in natural daily situations

such as playing, bathing, or sitting. In addition, suggestions are provided about various ways the

infant may be encouraged to explore the limits of its growing capacities. Interestingly, typical play

with siblings usually is an excellent means of promoting sensorimotor challenge, variation, and

exploration. At the same time, family members are informed about the basic principles of atypi-

cal development. The guidelines are: a) the notion that the appearance of motor behavior of the

child with special needs may differ from that of a typically developing child, but that this does not

matter as it is functional performance that counts; and b) the idea that development implies trial

and error during self-produced activity, in other words, that “error” production does not mean

failure but the presence of an active learning process, where learning may be enhanced by verbal

encouragement or non-verbal expressions of affection (e.g., hugging, touching, or tickling) by the

caregiver. The notions of NGST are not taught but discussed by means of bi-directional, equiva-

lent, and deliberate conversation, in line with the family involvement and educational component

of COPCA.

tIP based on NDt

TIP based on NDT is a multidisciplinary approach which primarily focuses on limitations

imposed by bodily impairments and functional activities of the infant with special needs. The as-

sumptions of the approach can be summarized with the help of these two major components:

1. Neurodevelopmental principles which consist of a mix of neuromaturational assumptions,

sensorimotor problem-solving strategies resembling those based on NGST, and the principles

of dynamic systems. The therapist plays a key role in the application of these principles due to

her/his instructional qualities of specific handling techniques and experiences. The therapist

teaches the infant to engage in developmental activities by providing sensorimotor experiences

accompanied by manual guidance, toys, a bolster, or a ball (Bly 1999, Howle 2002).

2. The family. The role of the family in the planning of the treatment program has been eloquent-

ly expressed by Bly (1999): “The family members are obviously the most important people on

the baby’s team” and “The more involved the family becomes, the more consistent therapeutic

management becomes for the baby” (p. 8).

The major aims of TIP based on NDT, which are listed below, are closely related to these two

components.

1. The neurodevelopmental component

In NDT the understanding of how typical developmental changes across a lifespan provides a

critical framework for the problem-solving approach and the planning of treatment. Development

is understood as an interplay between endogenous maturational processes and the infant’s own

environment. NDT uses problem solving to identify missing or atypical elements of functional

movements and posture in infants with a prenatally, perinatally, or early postnatally acquired le-

sion or malformation of the brain (Howle 2002).

The process of the guidance of an infant starts with the examination of functional limita-

tions. Next, on the basis of proposed relationships between limitations, intervention strategies are

systematically planned and applied. NDT emphasizes the individual as a whole with his/her own

competencies and limitations. The current enablement model of NDT addresses these competen-

cies and limitations – in line with the ICF-CY – in body, motor, individual, and social dimensions.

In infant treatment NDT emphasizes impairment as the starting point of therapeutic guidance

and improvement of impairment as a means to facilitate activity and participation (Bly 1999).

According to Mayston (2001) the basic philosophy underlying the sensorimotor deficits of the

infant with a brain lesion are pathophysiological insights into the mechanisms underlying the

problems in the coordination of posture and movement combined with atypical qualities of mus-

cle tone. The problems contribute directly to functional limitations. The impairments of function

can be classified into primary impairments, that is, those impairments that result directly from

the neuropathology along with secondary impairments. Two forms of primary impairments can

be distinguished: positive impairments such as spasticity and deficits in postural control, and

negative impairments such as muscle weakness and changes in muscle length. Those impairments

that are associated with atypical interactions within and between neural and body systems and

other environmental influences, such as contractures and atrophy, are classified as secondary im-

pairments (Howle 2002). For the implementation of NDT in daily practice this means that a broad

repertoire of tone-influencing facilitation techniques such as handling are used to reduce atypical

functional activities and to prepare the infant for optimally independent function.

Bly (1999) emphasized the importance of tactile, proprioceptive, kinesthetic, and vestibular

sensory input and the integration with the motor system in infant treatment based on NDT. The

sensory input enables the infant to perform movements and postural adjustments, and to learn

and use motor skills. Therefore, assessment of the infant must include attention to the sensory

system and treatment must include techniques that integrate sensory input with motor control.

The forms of NDT able to achieve this most precisely are the facilitation techniques of handling,

intermittent pressure, and tactile and vestibular stimulation. The hands of the therapist are used

to control sensory input, motor output, and muscle tone in such a way that the infant with neuro-

logical dysfunction gains an experience of typical movements and movement sequences with the

aim of enhancing the infant’s development of learning skills (Bly 1999).

At the same time the approach recognizes that typical development of postural control and

movements relies on the concept of dynamic systems (Thelen et al 1993). According to dynamic sys-

114 115

tems, motor development is brought about by self-organization; it is a result of interaction between

multiple factors such as body weight, muscle strength, joint configuration, the infant’s mood, and

the environment, including the characteristics of the task. These variables change during the devel-

opment of the infant, and NDT uses these variables to influence outcomes, for instance, by adapting

the intervention to the age of the infant and the infant’s characteristics in a direct teaching relation-

ship with the infant (Mayston 1992, Howle 2002). This implies that the therapist should treat the in-

fant and, during hands-on treatment, should select handling strategies that facilitate and prepare the

infant for age-specific function. The caregivers are instructed on how to continue and integrate these

treatment strategies which often continue to involve hands-on techniques throughout daily life.

2. The family involvement component

Therapists, other professionals, and the caregiver act like a team in the decision-making process

in order to establish the infant’s care plan. The role and responsibility of each discipline is de-

scribed in the treatment plan. This means that the therapy disciplines need to develop a treatment

plan in which the therapist and caregiver cooperate in the formulation of meaningful and relevant

functional goals. The therapist presents options and discusses these options with the parent in

order to reach a consensus, while anticipating the goals that the infant might achieve. The respon-

sibility of the therapist is to discover the best way for the infant to achieve his/her best potential.

This implies that the therapist should teach the child and train the caregivers and other family

members in the most useful ways of managing the infant (Bly 1999, Mayston 2001, Howle 2002).

The treatment plan incorporates caregiver education which is intended to facilitate the

caregiver-infant relationship, teach caregiver-specific skills, and enable the caregiver to handle/

assist with their child’s difficulties. This means that it is the responsibility of the caregiver to carry

out specific treatment strategies and to provide opportunities for practice. It is the therapist’s

role to guide the caregiver in how to modify the normal care-giving activities so that each daily

task can be used to reinforce the improvement of motor patterns which the infant has learned

during the therapy session. A greater involvement by the caregiver will express itself in terms of a

more consistent therapeutic management of the infant. In order to achieve an optimal result, the

therapist informs, instructs, and may give caregivers as many treatment ideas as the caregiver can

manage (Finnie 1996, Bly 1999). In addition, in TIP based on NDT, communication is regarded as

an essential means for service delivery. Communication is characterized by an open information

exchange between a source of information (the professional) and a receiver (the caregiver). In this

way information is exchanged about, for instance, specific details of the diagnosis, the child’s

prognosis, as well as on the presence or absence of physiological reactions, changes in muscle tone,

“why” and “how” to use handling techniques, along with communication skills (Finnie 1996).

The aim of the present study is twofold: 1) to examine whether it is possible to implement the con-

cepts of COPCA in daily physical therapy practice by specially trained pediatric physical therapists

and 2) to evaluate whether the content of COPCA sessions differs from TIP sessions.

table 6.1. Overview of differences between COPCA and TIP based on NDT.

COPCaFamily-relationship focused

tIP based on NDtChild focused

theoretical frame-works- Family involvement- Family education- Child development- Education of the infant

- Family autonomy (Law et al 1998)- Transactional model (Sameroff

and Mackenzie 2003) & ecological model (Bronfenbrenner 1967)

- NGST (Edelman 1989)- Individuality in parenting (Scarr

2001, Sanders et al 2004)

- Family centered care guided by profes-sional expertise (Howle 2002)

- Professional-client didactic relationship (Howle 2002, Mayston 1992)

- Mix of neuromaturation, dynamic systems and NGST (Howle 2002)

- General parenting principles (Finnie 1996)

Primary focus of guidance

- Decision making process of the family

- Optimizing child development

role of family - Key factor in process of coaching - Member of the team

role of therapist - Coach - Key person in guidance- Teacher

Education of infant - Coaching of caregiver - Educational perspective

- Therapist teaches infant and trains caregiver

Key words of motor learning

- Variation- Self-exploration- Challenge, testing the limits- Trial and error- Acceptance of atypical strategies

- Exposure to sensory-motor experience- Facilitation of typical motor behavior,

avoidance of atypical behavior

- Hands on hands off techniques

Communication - Bidirectional, equal partnership and open dialogue

- Open information exchange between a source of information (the professional) and a receiver (the caregiver).

116 117

Method

Subjects

Forty-six infants, who had been admitted to the Neonatal Intensive Care Unit (NICU) of the Uni-

versity Medical Center Groningen between March 2003 and May 2005 and who presented at the

age of ten weeks corrected age with definitely abnormal general movements (GMs), were included

in the study. GM assessment was carried out by MH-A and neonatologist AFB, who agreed on the

infant’s GM quality. The presence of definitely abnormal GMs indicates a high risk for develop-

mental disorder, including CP (Hadders-Algra 2004). Infants with severe congenital anomalies

and infants whose caregivers had insufficient understanding of the Dutch language were excluded

from the study. All caregivers of the infants signed an informed consent and the research project

was approved by the Ethics Committee of the University Medical Center Groningen. The trial was

registered under ISRCTN85728836.

Block randomization (full-term infants blocks of n=2; preterm infants blocks of n=12) alloca-

ted 21 infants to the experimental COPCA group and 25 infants to the control group. The infants

who were randomized into the control group received traditional infant physiotherapy (TIP) on

indication of the pediatrician. As a result, 22 infants were referred to TIP and three infants did not

receive physical therapy. Each group contained two full-term infants; the other infants had been

born preterm at gestational ages which varied from 25 to 36 weeks. Details of perinatal and social

characteristics of both groups are provided in Table 6.2.

The randomized intervention was provided at between 3 and 6 months corrected age (CA).

The COPCA sessions were performed twice a week for one hour in the home environment by

one of four COPCA coaches. The COPCA coaches were certified and registered pediatric physical

therapists who had received specific education in the COPCA program. The educational curricu-

lum of a COPCA course starts off with two days of training in the basic COPCA principles. These

two days include discussions of videos of recent intervention sessions carried out by participants.

Discussion of the videos allows for clarification of key characteristics of COPCA and the differ-

ences between COPCA and TIP. After the introductory two days, training is continued with four

one-day sessions with an interval of six weeks. During the interval, coaches-in-training videotape

their own intervention activities. Again, the videos are the starting point for discussion of how

the principles of COPCA may be applied in daily practice. The COPCA curriculum also involves

obligatory reading. During the study, COPCA coaches continued to receive supervision from the

first author. In earlier phases of their careers, three of the four COPCA coaches had completed the

NDT basic and infant course based on Neurodevelopmental Treatment (NDT) principles. Seven-

teen pediatric physical therapists were involved in the TIP treatment sessions. Sixteen of them

had completed the NDT basic and infant treatment course based on NDT. The frequency of TIP

sessions varied from two to 28 times (median 9), and their duration from 12 to 50 minutes (median

29). The great majority of TIP sessions were also carried out in the home environment (n=20; 91%)

Therapists for both groups had at least five years experience in treating infants and children with

special needs. None of the infants received additional forms of therapeutic guidance.

COPCa (n=21) tIP (n=25)Gender

Male

Female

n (%)n (%)

9 (43)12 (57)

11 (44)14 (56)

Gestational age at birth (wk) median (range) 29 (27-40)

30 (25-39)

Birth weight (g) median (range) 1210 (585-4750)

1143 (635-3460)

Maternal age mean ± SD 30.5 ± 6.2 31.8 ± 4.3

Firstborn child n (%) 12 (57) 13 (52)

Twin pairs n (%) 9 (43) 7 (28)

Abnormal cerebral ultrasound IVH gr. 3-4

PVL gr. 3-4

n (%)n (%)

1 (5) 2 (10)

1 (4) 1 (4)

Cerebral Palsy at 18 months n (%) 5 (24)

5 ( 20)

Maternal education * Low

Middle

High

n (%)n (%)n (%)

3 (14)16 (76) 2 (10)

3 (12)11 (44)11 (44)

* high vs middle + low education: Chi square: p<0.05 SD = standard deviation; wk = weeks; g = gram; gr = grade; IVH, grading according to Volpe (2000) = intraven-tricular haemorrage; PVL, grading according to de Vries et al. (1992) = Periventricular leukomalacia; Levels of education: low = primary education/ junior vocational training, middle = secondary education / senior vocational training, high = university education / vocational colleges.

Video recording and video analysis

At 4 and 6 months CA, a video recording of a physical therapy session was made. To minimize intru-

sion, the camera was positioned as far away from the therapist and infant as possible. Albrecht and

coworkers (2005) demonstrated that video recording scarcely affects the behavior of the people be-

ing filmed. In the COPCA group, one recording was missing at four months and three at six months

due to logistical difficulties. In the TIP group, one recording was missing at four months (logistics)

and one at six months (treatment had already stopped). This resulted in 41 video recordings at four

table 6.2. Perinatal and social characteristics of both study groups.

118 119

months (COPCA n=20, TIP n=21) and 39 recordings at six months (COPCA n=18, TIP n=21).

Two persons, CBH and LH, analyzed the contents of the sessions with a standardized observa-

tion protocol for the classification of PT actions (Blauw-Hospers et al 2010). Both assessors were

blind to group allocation. The analysis was performed with the Noldus software program “The

Observer” (Version 5.0; Noldus, Wageningen, the Netherlands), a program specifically designed for

behavioral observation. The program allows for the quantification of the duration, frequency, and

serial order of defined actions. The therapeutic actions defined in the protocol reflect the specific

components of COPCA and TIP (see Appendix 1, p. 188). The observation protocol classifies PT

actions into eight main categories: A) Family involvement and educational actions; B) Communi-

cation, C) Handling techniques; D) Sensory experience; E) Passive motor experience; F) Self-pro-

duced motor behavior, no interference; G) Challenge to self-produce motor behavior where infant

is allowed to continue activity; and H) Challenge to self-produce motor behavior that flows over

into therapeutic handling. In addition, the position of the infant and the amount of postural sup-

port the infant received during the PT actions was scored. The majority of outcome parameters

resulted in relative amounts of time spent on PT actions. Items from Sections 1 to 3 of Category A

(Family involvement and educational actions, A1 to A3) were categorical data. Categories A and B

describe aspects of family involvement and educational interaction in the intervention programs.

In Categories C to H, the position and postural support parameters deal with the neuromotor

components of treatment.

Previously we demonstrated that inter- and intra-assessor agreement on assessment with the

protocol were satisfactory. The ICCs on the relative duration of actions ranged from 0.76 to 1.00

for inter-assessor agreement and 0.69 to 0.99 for intra-assessor agreement (Blauw-Hospers et al

2010).

Data analysis

Statistical analyses were performed using the computer package SPSS (version 14.0). Non para-

metric statistics were used since none of the variables were normally distributed. The effect of

age was assessed with the Wilcoxon signed rank test. Differences between COPCA and TIP were

evaluated with the Mann-Whitney U test, the Chi-square test or Fisher Exact test. Differences

with a p-value <0.05 were considered statistically significant.

results

Family involvement and educational components of intervention

The caregivers were involved in both types of intervention, albeit in different ways. During TIP

sessions, caregivers were engaged especially in handling, and during COPCA, in playing (Table

6.3). The difference in approach between COPCA and TIP was also reflected in the way the child

was dressed: during TIP, infants were more often undressed than during COPCA, a difference

which reached statistical significance at four months (Table 6.3). In both types of intervention,

mothers were usually present during treatment sessions.

Intervention 4 months 6 monthsCOPCan= 20

tIPn= 21

COPCan= 18

tIPn= 21

Family involvementa.1 Family members involved in intervention session

Mother present only- 13 13 15 11Father present only- 1 1 2 0Both caregivers present, no other family members- 4 3 1 3In addition to parent(s) other family members - present 1

2 4 0 7

a.2 Role of parent, caregiver2

Physical therapist performs treatment by means of - handling

1 15 0 15

Physical therapist performs treatment (handling) - and parent guides attention of infant

0 5 0 5

Parent performs treatment by means of handling. - Physical therapist instructs how to handle

0 1 0 1

Parent and physical therapist act together (hands - off); parent is playing with the infant; physical the-rapist observes parent –infant relationship; parent gives ample opportunities for exploration

13 0 14 0

Parent is playing with infant (hands-off), ample op-- portunities for exploration

6 0 4 0

a.3 Infant dressing3

Dressed- 15 5 9 6Partially dressed- 4 5 6 5Undressed (wearing underwear only)- 1 11 3 10

1 In the current study other persons present were always grandparents. 2. Role of parent, caregiver: small (first three categories) vs large (last two categories). Chi Square p<0.001.3. Infant dressing: Chi Square for trend p<0.001.

table 6.3. Family involvement during COPCA and TIP sessions.

120 121

Inte

rven

tion

t

IPC

OPC

a

4 m

o6

mo

Pool

ed

4 an

d 6

mo

4 m

o6

mo

Pool

ed

4 an

d 6

mo

med

ra

nge

(%)

med

ra

nge

(%)

med

ra

nge

(%)

med

ra

n-ge

(%

)

med

ra

n-ge

(%

)

med

ra

nge

(%)

a. F

amil

y in

volv

emen

t and

Edu

cati

onal

co

mpo

nent

a.4

Edu

cati

onal

act

ion

s4.

1 1-

373.

8 1-

224.

0 1-

3717

.00

-44

12.7

0-3

616

.0**

0-4

4

- C

areg

iver

inte

rfer

es w

ith

in

fant

’s ac

tivi

ties

0.0

0-1

0.0

0-2

0.0

0-2

0.3

0-3

0.9

0-5

0.6*

*0

-5

-

PT

inte

rfer

es w

ith

infa

nt’s

acti

viti

es

2.1

0-3

22.

00

-17

2.1

0-3

20.

40

-80.

20

-70.

2**

0-8

-

PT

gui

des

the

infa

nt0.

00

-10

0.2

0-1

00.

2 0

-10

0.0

0-7

0.0

0-0

0.0*

*0

-7

- P

T g

ives

car

egiv

er t

rain

ing

0.1

0-1

30.

00

-50.

00

-13

0.0

0-3

0.0

0-1

0.0

0-3

-

PT

coa

ches

the

car

egiv

er0.

00

-10.

00

-10.

0 0

-112

.40

-43

11.3

0-3

511

.9**

0-4

3B

. Com

mun

icat

ion

10.5

0-5

515

.60

-45

12.4

0-5

517

.60

-50

14.7

2-52

15.7

0-5

2

- In

form

atio

n ex

chan

ge1.

60

-16

1.3

0-1

41.

40

-16

1.8

0-9

0.9

0-9

1.3

0-9

-

Con

tent

s of

info

rmat

ion

-

Dev

elop

men

tal e

duca

tion

0.7

0-2

01.

40

-31

1.3

0-3

11.

00

-20

2.1

0-3

11.

30

-31

-

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dlin

g0.

40

-10

0.3

0-1

40.

4 0

-14

0.0

0-1

0.0

0-0

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*0

-0

- Va

riat

ion

0.0

0-1

0.3

0-1

40.

0 0

-11.

60

-90.

50

-81.

3**

0-9

0

- A

DL

hand

ling

0.0

0-1

00.

00

-50.

0 0

-10

0.0

0-0

0.0

0-0

0.0*

*0

-0

- A

DL

vari

atio

n0.

00

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00

-00.

0 0

-00.

0

0

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00

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0**

0-7

-

Inst

ruct

-

Ass

ign

0.2

0-3

0.2

0-3

0.2

0-3

0.9

0-1

21.

00

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0**

0-1

2

- G

ive

hint

s0.

00

-40.

00

-10.

0 0

-42.

50

-11

2.1

0-5

2.3*

*0

-11

-

Pro

vide

feed

back

3.5

0-1

73.

10

-11

3.3

0-1

7 3

.70

-14

5.1

0-1

34.

50

-14

-

Impa

rt k

now

ledg

e2.

30

-17

1.4

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72.

80

-14

1.7

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-14

Neu

rom

otor

com

pone

ntC

. Fac

ilit

atio

n te

chn

ique

s 31

.75-

7428

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5428

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5-74

2.6

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30

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2.9*

*0

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- Han

dlin

g 16

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5716

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4216

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3-57

0.4

0-3

20.

30

-40.

4**

0-3

2- P

ress

ure

tech

niq

ues

6.3

0-2

55.

30

-24

6.2

0-2

50.

00

-50.

00

-80.

0**

0-8

- Sup

port

dev

ice

0.0

0-2

50.

00

-15

0.0

0-2

50.

00

-51.

80

-50.

0**

0-5

- Tra

nsi

tion

3.6

0-1

74.

01-

163.

8 0

-17

1.8

0-5

0.0

0-1

1.8*

*0

-5D

. Sen

sory

exp

erie

nce

8.5

1-21

6.1

0-3

66.

1 0

-36

1.9

0-1

11.7

0-7

1.8*

*0

-11

E. P

assi

ve m

otor

exp

erie

nce

4.6

0-2

02.

00

-19

2.4

0-2

00.

00

-40.

00

-10.

0**

0-4

4F.

Sel

f-pr

oduc

ed m

otor

beh

avio

r –

no

inte

rfer

ence

21

.30

-43

21.3

1-83

21.3

0-8

141

.02-

7152

.57-

6944

.3**

2-71

G. C

halle

nge

to s

elf-

prod

uced

mot

or b

e-ha

vior

– a

ctio

n co

ntin

ued

by in

fant

15.9

0-3

212

.32-

3813

.2

0-3

835

.23-

8130

.817

-70

31.0

**3-

81

- Lit

tle

vari

atio

n12

.40

-28

8.3

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010

.70

-34

2.4

0-4

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50

-41

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ge v

aria

tion

1.3

0-1

60.

00

-27

1.0

0-2

726

.52-

5925

.57-

3725

.8**

2-59

H. C

halle

nge

to s

elf-

prod

uced

mot

or b

e-ha

vior

– w

ith

over

flow

into

han

dlin

g5.

51-

255.

61-

375.

6 0

-37

0.0

0-6

0.0

0-5

0.0*

*0

-6

- Lit

tle

vari

atio

n4.

90

-23

3.7

0-3

74.

9 0

-37

0.0

0-6

0.0

0-1

0.0*

*0

-6- L

arge

var

iati

on0.

00

-25

0.0

0-6

0.0

0-2

50.

00

-10.

00

-40.

00

-4

I. U

nsp

ecifi

ed a

ctio

ns

1.9

0-1

33.

50

-11

3.2

0-1

32.

91-

93.

11-

93.

11-

9

Bol

d in

dica

tes

a si

gnifi

cant

age

-eff

ect,

Man

n W

hitn

ey, p

< 0

.05.

Diff

eren

ces

betw

een

TIP

and

CO

PCA

**

p <

0.01

A

DL

= ac

tivi

ties

of d

aily

life

, Med

= M

edia

n, M

o =

mon

ths

(cor

rect

ed a

ge),

PT

= p

hysi

cal t

hera

pist

.

tabl

e 6.

4. C

onte

nts

of T

IP a

nd C

OPC

A in

terv

enti

on a

t 4 a

nd 6

mon

ths

corr

ecte

d ag

e: p

ropo

rtio

n of

tim

e sp

ent o

n va

riou

s ac

tion

s.

122 123

The time spent on various educational actions varied widely (Table 6.4 and Figure 6.1). Age had

only a minimal effect on family involvement and educational actions; we therefore pooled the

four- and six-month data for the evaluation of the differences between COPCA and TIP (Table

6.4). During COPCA sessions more time was spent on educational actions (median value 16%) than

during TIP sessions (median value 4%, p<0.001). Not only did the duration of educational actions

differ between the two treatments, but the contents also did. During TIP sessions, educational

actions more often consisted of actions during which the therapist interfered with the infant’s ac-

tivities, that is, corrected, interrupted, or assisted infant activities. During COPCA sessions, most

of the time was spent on coaching the caregivers, whereas this form of guidance did not occur in

the TIP group (p< 0.001).

In both groups an equal amount of time was spent on communication. However, differences were

present in the contents of the communication, especially in the type of information provided by the

therapist (Table 6.4 and Figure 6.1). During TIP sessions, the focus was on explaining facilitation and

the use of handling techniques in terms of hand placing, tone influencing, asymmetry, symmetry,

and typical movement patterns. During COPCA sessions, the emphasis was on explaining the impor-

tance of variation and the role of minimal support, exploration, and trial and error. COPCA coaches

more often used the instructional strategy of “giving opportunities” than did TIP therapists.

0 20 40 60 80

PT coaches parent

Interruption infant behavior

Communication

Handling techniques

Sensory experience

Passive experience

CSMB*followed by handling

CSMB* continued by infant

Time spent on PT actions (%)

tIPCOPCa

Figure 6.1: Data on the relative duration of time spent on eight categories of PT actions during treatment sessions. The horizontal lines represent ranges; the boxes, the interquartile ranges; and the vertical bold bars, median values. CSMB = Challenged Spontaneous Motor Behavior

Neuromotor components of intervention

The application of physical therapy at four and six months was characterized by heterogeneity

(Table 6.4). The relative duration of the majority of PT actions in both groups at four months did

not differ significantly from that at six months (Table 6.4). The only exception to this rule was

a slight increase for “Challenge to self-produce motor behavior with overflow into therapeutic

handling” in the TIP group from four to six months. In light of the minimal effect of age, we

considered it justified to pool the data of four and six months in our evaluation of the differences

between COPCA and TIP (Table 6.4 and Figure 6.1).

During TIP sessions most of the time was spent on facilitation (median value 28%), especially

on handling. Handling techniques were almost absent in the COPCA group, meaning that the

difference in time spent on handling techniques between TIP and COPCA sessions was highly

significant (p<0.001). During TIP sessions a substantial amount of time was also spent on granting

the infant time to produce motor behavior by itself (either entirely spontaneously: 21%; challenged:

13%). During COPCA sessions even more time was spent on granting the infant time to produce

motor behavior by itself, either entirely spontaneously (44%; difference with TIP: p<0.001) or

challenged (31%; difference with TIP: p<0.001). We noted also a qualitative difference between the

two groups during the periods of entirely spontaneous activity. In the TIP group, this meant that

the infant was left unattended while therapist and caregivers were engaged in communication; in

the COPCA group, this meant that caregivers and PT observed and discussed the infant’s activi-

ties. When the infants during COPCA sessions were challenged by toys or the face of the physical

therapist or caregiver to explore their own movement possibilities, considerably more variation

was used than during TIP sessions (median values COPCA 26% vs. TIP 1%, p<0.001). During

COPCA sessions little time was spent on sensory stimulation, passive experiences, and challeng-

ing of motor behavior flowing over into handling techniques; these actions occurred significantly

less often than during TIP sessions (Table 6.4 and Figure 6.1).

The position of the infant during treatment varied with treatment type and age. With increas-

ing age, infants were less frequently treated in supine position and more often in prone and sitting

positions (Table 6.5). During TIP sessions, infants were handled significantly more often in supine

position with the pelvis slightly lifted and in prone than during COPCA sessions; this was true for

both ages. During COPCA sessions, infants were more often placed in sitting position, particular-

ly at four months. It is well known that young infants need postural support during sitting. It was

therefore interesting to note that during COPCA more time was especially spent on sitting with

minimal support (Table 6.5).

124 125

Inte

rven

tion

tIP

CO

PCa

4 m

o6

mo

4 m

o6

mo

Med

ian

R

ange

(%)

Med

ian

R

ange

(%)

Eff

ect

age;

P-va

lue

Med

ian

R

ange

(%

) M

edia

n

Ran

ge

(%)

Eff

ect

age;

P-va

lue

Posi

tion

Su

pine

pel

vis

not l

ifte

d 38

.92-

6627

.95-

600.

06 4

5.6

7-92

34.3

0-5

50.

68Su

pine

wit

h pe

lvis

slig

htly

lift

ed16

.50

-47

9.5

0-3

20.

210.

0 **

0-2

70.

0 **

0- 7

0.06

Pron

e15

.67-

3529

.412

-43

0.00

25.

6 **

0-2

914

.2 *

0-3

80.

008

Side

5.6

0-4

34.

00

-15

0.20

3.8

0-2

75.

50

-33

0.95

Sitt

ing

14.5

3-83

25.3

9-50

0.00

236

.7 *

*0

-72

44.9

**

5-85

0.28

- Sit

ting

wit

h no

sup

port

0.0

0-0

0.0

0-2

0.04

70.

00

- 50.

4 *

0-1

50.

002

- Sit

ting

wit

h m

inim

al s

uppo

rt0.

30

-93.

50

-32

0.00

0513

.6 *

*0

-42

19.8

**

1-48

0.14

- Sit

ting

wit

h cl

ear

supp

ort

5.9

0-3

211

.60

-41

0.78

17.6

*0

-38

10.2

1-26

0.23

- Sit

ting

wit

h fu

ll su

ppor

t7.

00

-42

4.0

0-3

20.

591.

40

-25

1.4

0-3

4

0.52

Bol

d in

dica

tes

a si

gnifi

cant

age

-eff

ect,

Man

n-W

hitn

ey p

< 0.

05. D

iffer

ence

s be

twee

n C

OPC

A a

nd T

IP:

* p<

0.0

5, *

* p<

0.0

1M

o =

mon

ths

(cor

rect

ed a

ge)

tab

le 6

.5. I

nfa

nt p

osit

ion

and

degr

ee o

f pos

tura

l sup

port

in s

itti

ng d

urin

g T

IP a

nd C

OPC

A s

essi

ons:

pro

port

ion

of t

ime

spen

d in

var

ious

pos

itio

ns.

Discussion

The present study demonstrated that the contents of COPCA and TIP sessions differed widely,

suggesting that it is possible to implement COPCA in daily practice by pediatric physical thera-

pists who have received special training in the COPCA program. The differences found cor-

respond to the theoretical frameworks of the two approaches. They included differences in the

approach to families, the role of the caregiver and pediatric physical therapist, the application of

educational actions towards the infant, and in activities to stimulate the infant’s sensorimotor

development.

Physiotherapeutical considerations

Our data clearly demonstrated that, notwithstanding the fact that both TIP and COPCA are

defined as family-centered services (Rosenbaum 2004), important differences between the two

approaches exist in application of the key element “family involvement.” The videos of the COPCA

sessions illustrated that family members, including the infant with special needs, were the key

persons in guidance which consisted of coaching. Caregivers determined the focus of the guid-

ance. The COPCA coaches created a situation in which caregivers felt free to explore and discuss

alternative strategies. Caregivers took care of or played with the infant (in a hands-off manner) to

provide the infant with ample opportunities for self-exploration. In contrast, TIP therapists usu-

ally treated the infant by using handling techniques (hands-on/hands-off) and provided parents

with references about what the therapist was doing or what a parent could do in daily practice. It

was also observed that TIP therapists were often in charge of decision-making. This was reflected,

for instance, in the way they taught infants the “best” way to accomplish self-initiated movement

goals. The observed characteristics of TIP correspond with the descriptions of baby treatment

based on NDT principles (Bly 1999).

The differences between COPCA and TIP reflect a profound difference in the role of the family

in the decision-making processes involved in the care of a child with special needs. During the

past few decades, understanding of how to manage the care of children with special needs has

changed substantially. This has resulted in the advocacy of a family-centered instead of a child-

focused approach (Rosenbaum 2004). Our data on the daily practice of TIP therapists indicate

that therapeutic guidance in general is still characterized by child-focused care with a unilateral

dominance of the professional determining the goals of and actions in treatment. Thus the TIP

approach corresponds to the direct didactic teaching principles of NDT, put into words by May-

ston (1992) in the following way (p. 6): “Our responsibility as therapist is to discover the best way

for our clients to achieve their best potential.” The discrepancy between current theory (family

focus) and daily practice is in line with the findings of O’Neil and Palisano (2000) which indicate

that therapists who acknowledged the importance of family-centered care focused their profes-

sional attention on the child’s impairment. We suggest that the critical determinant for family-

centered services, as suggested by COPCA, is family autonomy. This implies that professionals

should step back and restrict their role to coaching. The COPCA coach helps caregivers to discover

126 127

and develop their own problem-solving strategies for coping with and caring for the infant with

special needs. The important distinctions between COPCA and TIP are that 1) in COPCA the role

of the family is not restricted to parental involvement in the decision-making in terms of func-

tional treatment goals, and 2) COPCA refrains from teaching caregiver intervention techniques

(Bromwich 1976, Affleck 1982, Mahoney 1998).

The differences observed in activities to stimulate the infants’ sensorimotor development

between the two groups correspond to the theoretical frameworks of the two approaches. The

videos demonstrated that the COPCA approach creates a rich and varied world of opportunities

allowing the infant to explore variable motor behavior (hands-off). A case in point is the challenge

of postural behavior by the provision of as little support as possible. By creating a challenging en-

vironment, the infant may, through active trial and error, experience which motor behavior is the

best he or she can manage. This corresponds to the ideas of NGST. Preliminary results of a study

on the effect of COPCA and TIP on postural development in early infancy suggest that COPCA

may enhance the selection of a functional postural strategy (De Graaf-Peters et al 2007). The TIP

approach also aims at facilitating motor behavior produced by the infant her/himself; however,

our study indicated that TIP therapists frequently incorporated techniques of handling and care-

fully graded stimulation (hands-on). Therapists in the TIP group also applied the basic concept of

NDT to prepare the infant’s motor capacities for a specific function (Mayston 1992). This was, for

instance, reflected in the use of treatment in supine position with the pelvis slightly lifted during

TIP sessions, whereas this type of positioning was virtually absent during COPCA sessions. This

specific handling technique aims to facilitate functional activities such as rolling or reaching and

grasping in the midline, while the therapist controls muscle tone. It should be noted, however,

that the question of how increased muscle tone is related to activity limitations and postural

control is a matter of debate (Damiano 2006, Brogren-Carlberg and Bower 2008). Analogous to

the findings in the family-care domain, our findings in the neuromotor domain match those of

O’Neil and Palissano (2000): Despite the presence of a shift to more functional therapy, therapists

generally pay more attention to movement quality than to functionality so as to achieve a better

long-term outcome. This suggests that the “living concept” approach of NDT – as recognized by

senior NDT tutors – may benefit from a paradigm shift from “quality” (how) to “functionality”

(what) (Mayston 1992).

Methodological considerations

The major strength of the study is the standardized, video-based analysis of the contents of the

two forms of infant physical therapy that allows for an objective quantification of differences

between the two approaches. Other strengths of the study lie in the identical age of the infants at

inclusion in the study, the identical ages at video-assessment, and identical limitations at study

entry, that is, the presence of definitely abnormal GMs. The presence of definitely abnormal GMs

at three months post-term is associated with a high risk of developmental motor disorders such as

cerebral palsy (Prechtl 2001, Hadders-Algra 2004).

It may be considered a weakness that the therapists who applied COPCA had received a specific

COPCA training shortly before the study started, whereas training of the therapists who applied

TIP had taken place at longer and more variable periods prior to study onset. Nevertheless, the

great majority of TIP therapists received certified NDT training including an infant treatment

course. It is well known that therapists practicing NDT, develop a personal, eclectic approach as

their experience increases (Mayston 2008). This was reflected in the variation in percentage of

time spent on specific actions during the treatment sessions – a variation which partly blurred

the contrast between TIP and COPCA. Yet, by using a control group of therapists applying regular

TIP, we were able to demonstrate that the contents of COPCA sessions differed from currently

practiced infant physiotherapy in the Netherlands.

The difference in treatment frequency may be regarded as another limitation, as frequency of

treatment has been associated with outcome (Tsorlakis et al 2004).

At the time of the study, the current design had the highest level of feasibility. In our analysis

of the effect of intervention, we took frequency of treatment into account (Blauw-Hospers et al,

submitted for publication).

Infant physical therapy differs across countries and varies with the cultural context of fami-

lies. This means that the findings of the present Dutch study cannot immediately be generalized

to other countries.

The randomized design of the study was associated with a high degree of similarity for both

groups of infants (Table 6.2). Despite randomization, the level of maternal education differed be-

tween the two groups. This difference may be considered a weakness of the study, since contents

of a treatment session may be affected by the level of maternal education.

Concluding remarks

Bertha and Karl Bobath were well aware of the possibility of future progress in ideas on family

care and motor development. They encouraged therapists to incorporate new ideas into daily ap-

plication of therapy. In a way, one could say that the authors of the COPCA program followed the

advice of the Bobaths. They developed a new program, COPCA. The current study indicates that

COPCA and TIP based on NDT differ widely, justifying the label “novel program.”

Our study underscores the notion that the application of standardized, computer-based video

analysis of treatment sessions is an invaluable tool in the understanding of the daily practice of

pediatric physical therapy. Evaluation of the videos taught us that physical therapists often do

other things than they intended to do (Blauw-Hospers et al 2010). This means that only the evalu-

ation of real action and communication can provide insight into the reality of practice.

128 129

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C H a P t E r 7

P e dI At r Ic P h ys Ic A l

t h e r A P y I n I n fA nc y:

f rom n Ig h t m A r e to

dr e A m ?

A r A n d om I z e d

c on t rol l e d t r I A l ---------------------------------------------------------------------------------------------

Cornill H. Blauw-Hospers MSc, Tineke Dirks PT, Lily J. Hulshof MD,

Arend F. Bos MD, PhD and Mijna Hadders-Algra MD, PhD

Submitted for publication

132 133

abstract

Purpose. To study the effects of a new pediatric physiotherapeutic intervention program on the

development of infants at high risk for developmental disorders. We used a combined approach in

a randomized controlled trial (RCT) and process evaluation.

Subjects and Methods. Forty-six high-risk infants were randomly assigned to receive COPCA

(n=21) or traditional infant physical therapy (TIP; n=25) between three to six months corrected

age (CA). Developmental outcome was assessed at six and 18 months CA with a neurological

examination, AIMS, PEDI, and the mental developmental index (MDI) of the BSID. Contents of

the intervention were analyzed by a quantitative video analysis of therapy sessions. Multivariate

statistics were applied to evaluate associations between intervention and developmental outcome

components.

results. The RCT revealed that developmental outcome in both groups was largely identical, ex-

cept for cognitive outcome. The COPCA group decreased less in MDI score from six to 18 months

CA (106.5 to 100; p=0.07) than the TIP group (115.5 to 98; p=0.001). Process evaluation showed that

typical COPCA actions – 1) family involvement and educational actions, 2) application of a wide

variation in challenging the infant to produce motor behavior by him/herself and allowing the

infant to continue this activity, and 3) stimulation of motor behavior at the limit of the infant’s

capabilities – were associated with a better developmental outcome at 18 months CA. The use of

handling techniques was associated with a lower score on the PEDI.

Conclusion. Extending the RCT by using process evaluation was needed to obtain insight into

associations between the components of intervention and developmental outcome. Specific

components of COPCA were associated with improved outcome, while handling techniques were

associated with worsened functioning.

Introduction

Research has shown that evidence for the effectiveness of pediatric physical therapy on motor and

cognitive development of infants with or at high risk for developmental disorders is inconclusive

(Blauw-Hospers and Hadders-Algra 2005, Blauw-Hospers et al 2007, Spittle et al 2007). However,

early pediatric physical therapy is widely advocated and desired in the management of high-risk

infants. This inconclusiveness may be related to the specific difficulties inherent in studies on

early pediatric physical therapeutic intervention. One important difficulty for these studies is the

heterogeneity of the study group. Early detection of infants at risk for a developmental disorder

implies that the clinical picture of the problems that an infant will eventually develop has not yet

manifested itself. Second, there are many variations in the conditions of families such as social-

economic status and family routines. Another, equally important factor that might explain the

inconclusiveness of results is related to the intervention itself. Treatment application is often ec-

lectic (Howle 2005, Mayston 2005). This would indicate that a broad heterogeneity in implement-

ing a treatment exists, even within treatments that adhere to the same principles (Blauw-Hospers

et al 2010). Pediatric physical therapists tend to include in their treatment those techniques which

they perceive as helpful, while leaving other techniques out. Presumably this heterogeneity in

treatment practice has been brought about by an evolution in treatment techniques and theoreti-

cal assumptions. Finally, the outcome measurements that are chosen to evaluate the effect of an

intervention might not be the appropriate ones. Usually instruments are chosen out of habit, as

well as for practical reasons, and not on the basis of information regarding test accuracy, utility,

and theoretical basis (Ketelaar et al 1998, Heineman and Hadders-Algra 2008).

The best method for controlling the impact of the above-mentioned factors is to evaluate the

effect of intervention in a randomized controlled trial (RCT). RCTs are offered as the most objec-

tive and unbiased design for evaluating intervention programs (Lachin et al 1988). Random alloca-

tion of intervention among the participants ensures that differences in group characteristics that

may affect outcome are minimized. The aim is to have the groups compared as similarly as pos-

sible except for the precisely defined interventions being examined. As in other research designs,

RCTs in pediatric physical therapy also have limitations. RCTs are usually designed to determine

the effect of intervention by measuring the change in developmental outcome previous to and

after the intervention period. Although this is the most straightforward method for measuring ef-

fectiveness, it does not explain the underlying mechanisms that might influence outcome (Watson

et al 2004). Rarely are the contents of pediatric physical therapy programs analyzed despite the

fact that this might serve as an eye-opener in interpreting the results of effectiveness studies. Un-

raveling the components of intervention shows whether the applied intervention is eclectic or not.

Lettinga et al. (2002) stressed the importance of an in-depth understanding of the characteristics

of interventions. They stated that every trial which compares the effects of different types of in-

tervention should start with a detailed analysis of the similarities and differences in the content of

the interventions under study. This analysis will serve as an addition to discovering the effective

and ineffective elements in the interventions, and will consequently result in knowledge about the

134 135

implementation of the intervention by different pediatric physical therapists in different settings.

A second limitation that an RCT has is related to the validity of using experimental methods in re-

search addressing human activity, which is, for example, the case in early intervention programs.

Watson et al. (2004) argue that changes in behavior and psychosocial issues are better evaluated

with qualitative approaches, even though these approaches may be less robust.

This paper will describe the results of an RCT on pediatric physical therapy in infancy. In an

early intervention project, the VIP project (VIP is the Dutch abbreviation for “Vroege Interventie

Project”), we studied the effects of early pediatric physical therapeutic intervention on the devel-

opmental outcome of infants with or at high risk for developmental disorders. The effects of a

recently developed early intervention program, COPCA (COPing with and CAring for infants with

special needs: a family-centered program; Dirks and Hadders-Algra 2003, Dirks et al, submit-

ted for publication), were compared to the effects of traditional infant physical therapy (TIP). To

overcome the aforementioned limitations of RCTs, we extended our RCT with a process evalua-

tion to examine how COPCA and TIP were implemented in daily pediatric physical therapeutic

practice. Based on the contents of both interventions, we attempted to identify whether changes

in developmental outcome were associated with the contents of the intervention or whether they

could be explained by other factors. To document developmental changes, a set of instruments

was used, ranging from instruments measuring at impairment level (neurological examination) to

instruments at the level of activity and participation (PEDI).

Method

Participants

The study groups of the VIP project consisted of infants who had been admitted to the neonatal

intensive care unit of the University Medical Center Groningen between March 2003 and May

2005. At ten weeks corrected age, a video recording was made of the infants’ General Movements

(GMs). Infants and their families were selected to participate in the project when the quality of

the infants’ GMs was classified as definitely abnormal. The presence of definitely abnormal GMs

at fidgety GM age (i.e., at 2-4 months post-term) indicates a high risk for developmental disabili-

ties, such as cerebral palsy (CP) (Prechtl 2001, Hadders-Algra 2004). Infants with severe congeni-

tal anomalies and infants whose caregivers had an inappropriate understanding of the Dutch

language were excluded from the study. Informed consent was obtained and the research project

was approved by the Ethics Committee of the University Medical Center Groningen. The trial is

registered under ISRCTN85728836.

Forty-six infants participated in the VIP project. Through block randomization (full-term

infants blocks of n=2; preterm infants blocks of n=12) the infants were assigned to COPCA (n=21)

or TIP (n=25). The groups were comparable on baseline characteristics, such as gender, gestational

age, birth weight, and presence and severity of brain lesions (Table 7.1), but the groups differed for

maternal education, which was significantly better in the TIP group (Table 7.1). The randomized

intervention was provided between three and six months corrected age (CA). After the interven-

tion period, pediatric physical therapy was only continued when the pediatrician in charge of the

infant considered it necessary.

* p<0.05SD = standard deviation; wk = weeks; g = gram; gr = grade; IVH, grading according to Volpe (2000) = intra-ventricular haemorrage; PVL, grading according to de Vries et al (1992) = Periventricular leukomalacia; Levels of education: low = primary education/ junior vocational training, middle = secondary education / senior vocational training, high = university education / vocational colleges

COPCA (n=21) TIP (n=25)

Gender Male

Female

n (%)9 (43)12 (57)

11 (44)14 (56)

Gestational age (wk) median (range) 29 (27-40)

30 (25-39)

Birth weight (g) median (range) 1210 (585-4750)

1143 (635-3460)

Maternal age mean ± SD 30.5 ± 6.2 31.8 ± 4.3Firstborn child n (%) 12 (57) 13 (52)Twin pairs n (%) 9 (43) 7 (28)Abnormal cerebral ultrasound

IVH gr. 3-4

PVL gr. 3-4

n (%)1 (5)2 (10)

1 (4)1 (4)

Maternal education * Low

Middle

High

n (%)3 (14)16 (76)2 (10)

3 (12)11 (44)11 (44)

Intervention

TIP

TIP consisted of infant physical therapy as it is applied nowadays in the Netherlands. For the

most part it consists of the implementation of the “living concept” of neurodevelopmental treat-

ment (NDT) principles which primarily focus on the sensorimotor functional problems of the

infant (Howle 2002, Dirks et al, submitted for publication). More recently, influences from a more

functional approach, such as the encouragement of self-produced motor behavior, have also been

incorporated into the treatment. The functional approach emphasizes that critical parts of motor

learning are finding solutions for new task demands and adaptation to changes in the environ-

mental context (Ketelaar et al 2001, Ekström et al 2005). Due to the different influences which

table 7.1. Baseline characteristics.

136 137

have been incorporated into TIP, a broad heterogeneity in treatment applications was expected. In

the TIP group, intervention was applied with a median value of once a week, mainly in the home

environment by the pediatric physical therapist working in the area.

COPCA

COPCA (COPing with and CAring for infants with special needs: a family-centered program)

(Dirks and Hadders-Algra 2003, Dirks et al, submitted for publication) was developed from the

notion that evidence for the effectiveness of the current pediatric physical therapeutic interven-

tion programs was inconsistent. COPCA differs from existing approaches, both in theoretical

background and in implementation (Dirks et al, submitted for publication). It is a family-rela-

tionship-oriented program consisting of two theoretical components. The first is formed by a

family-involvement and educational-parenting component. A fundamental idea of COPCA is the

transactional perspective on family function, in other words, on patterns of reciprocal contingent

interactions between caregiver and infant, with the notion that changing patterns of behavior

are a characteristic of early development (Fiese 1997, De Weerth and Van Geert 2002). This means

that development is seen as a result of a continuous dynamic interplay between child behavior and

caregiver responses to the child’s behavior, along with environmental variables that may influence

both child and caregiver. Caregivers are coached to recognize the infant’s signals and to respond

appropriately to the actual needs of the infant (Dirks et al, submitted for publication). Coaching

implies that the pediatric physical therapist – in the COPCA program, the coach – supports all

family members, including the infant with special needs, in order to reveal their competencies,

goals, desires, and hopes. On the basis of an ongoing equal partnership in which the family defines

the priorities for intervention (Law et al 1998), the coach supports the family in developing their

own ways of caring for the infant and in improving personal coping skills. Specific attention is

paid to educational actions such as spending brief amounts of time playing in child-preferred

activities whereby the motor principles of NGST are taken into account, as well as educational ac-

tions promoting appropriate behavior (Dirks et al, submitted for publication).

The second, equally important, theoretical component of COPCA is a neurodevelopmental

component based on the principles of Neuronal Group Selection Theory (NGST; Edelman 1993).

NGST emphasizes that development is the consequence of a complex interaction between genetic

information and environmental influences. According to NGST, development is characterized by

two phases of variability, that is, primary and secondary variability. During primary variability the

child explores all the variations of motor possibilities that are available in the nervous system. In

this phase the child is not yet able to adjust his behavior to external conditions. In typically devel-

oping infants, this phase is characterized by abundant variation. At function-specific ages the in-

fant reaches the phase of secondary variability, that is, the child gradually learns to select the most

efficient solution for a given task out of his motor repertoire. This selection is based on self-pro-

duced trial and error experiences (Hadders-Algra 2000a). Infants with a prenatally, perinatally, or

early postnatally acquired lesion or malformation of the brain have a reduced repertoire of motor

strategies available for exploration. This is already expressed during the initial postnatal months

by a limited repertoire of general movements and continues when goal-directed motility emerges

(Hadders-Algra 2000b, 2008a). In addition, these infants have problems with the selection of the

most appropriate solution for a certain task out of the repertoire due to deficits in the processing

of sensory information (Hadders-Algra 2000b, 2008a). The aim of COPCA is therefore to promote

self-produced motor behavior (hands-off), variation, and trial-and-error experiences by means of

play, all with the ultimate goal of providing an infant with a wider diversity in terms of neuronal

networks that will help the infant to find an appropriate solution for any given motor task.

COPCA was applied twice a week for one hour at home by one of four specially trained

pediatric physical therapists.

Developmental outcome

The developmental status of the infants was assessed at baseline three months CA and thereafter

at six and 18 months CA. At three and six months, the neurological condition was measured with

the Touwen Infant Neurological Examination (TINE; Hadders-Algra et al 2010). The neurologi-

cal condition was summarized as normal, normal-suboptimal, minor neurological dysfunction

(MND), or as abnormal. The classification of abnormal during early infancy implies the presence

of a distinct neurological syndrome such as a clear hypo- or hypertonia, a hemi syndrome, or a

hyperexcitability syndrome. To distinguish between MND, normal-suboptimal, and normal neu-

rological conditions, the findings of the TINE were classified according to age-specific norms into

five clusters of dysfunction: dysfunctional reaching and grasping, dysfunctional gross motor func-

tion, brain stem dysfunction, visuomotor dysfunction, and sensorimotor dysfunction. Two forms

of normal neurological development could be distinguished: neurologically normal when none of

the clusters met the criteria for dysfunction and normal-suboptimal when one or two clusters ful-

filled the criteria for dysfunction. When more than two clusters fulfilled the criteria for dysfunc-

tion, infants were classified as MND. A recent study has shown that MND can be assessed reliably

with the TINE (Hadders-Algra et al 2010).

At 18 months CA, a neurological assessment was carried out according to Hempel (1993).

The findings of the Hempel assessment were classified as neurologically normal, simple MND,

complex MND, or CP. The distinction between simple and complex MND was also based on the

number of clusters of dysfunction. Note that the clusters of dysfunction of the Hempel assess-

ment are similar to that of TINE but not identical. The criteria for classification also differed for

both assessments. Simple MND at 18 months denotes the presence of one cluster of dysfunction

and complex MND the presence of more than one cluster of dysfunction. The classification CP

implies the presence of a “classical” configuration of neurological signs (Hadders-Algra 2003). Lit-

tle information was available about the concurrent and predictive validity of the Hempel assess-

ment, but the interrater reliability was satisfactory (Hadders-Algra et al 2010).

Additionally, we used the Neurological Optimality Score (NOS; Huisman et al 1995) to sum-

marize neurological condition at 18 months CA. The NOS is the sum of 57 items representing the

138 139

neurological examination that meets predefined criteria for optimality. It is important to realize

that the definition of optimal is narrower than that of normal or typical, and that a reduced

optimality does not always mean abnormal (Prechtl 1980). The NOS has been proven to be an

excellent instrument for evaluating subtle differences in neurodevelopmental outcome (Bouwstra

et al 2006).

Gross motor development was assessed with the Alberta Infant Motor Scale (AIMS; Piper and

Darrah 1994). The AIMS consists of 58 items that evaluate gross motor function in supine, prone,

sitting, and standing positions. Through observation of spontaneous motor behavior, each item

can be scored on three aspects of motor performance: weight bearing, posture, and antigravity

movements. The AIMS has very good reliability coefficients (Piper and Darrah 1994, Heineman

and Hadders-Algra 2008). In addition, the validity of the AIMS has been thoroughly examined and

proven to be satisfactory (Piper and Darrah 1994, Darrah et al 1998, Liao and Campbell 2004).

The cognitive outcome of the infants was assessed at six and 18 months CA using the men-

tal scale of the Dutch version of the Bayley Scales of Infant Development (BSID-II; Bayley 1993,

Van der Meulen et al 2002). The mental developmental index (MDI) consists of items concerning

problem solving, memory, discrimination, classification, language, and social skills. Raw scores

were converted into age-equivalent scores, as derived from the Dutch norms (Van der Meulen et

al 2002). The interrater reliability of the MDI was sufficient; the construct and concurrent validity

were moderate (Heineman and Hadders-Algra 2008).

The Pediatric Evaluation of Disability Inventory (PEDI; Haley et al 1992) was used to measure

the functional ability of the child. The PEDI was developed for young children from six months to

7.5 years of age and adapted to a Dutch version by Custers et al. (2000a) The PEDI is a discrimina-

tive measurement which aims to detect whether a child has limitations in his functional status

and, if so, to determine the extent and content area of the limitations. With the PEDI, both the

capability of the child (what a child can do) and the performance (what the child actually does)

of routine daily childhood activities can be evaluated. Capability is measured by the functional

skills scale, while the caregiver assistance scale and the modifications scale provide information

on the performance. Each scale consists of three domains, namely, self-care, mobility, and social

function. The PEDI can be considered a reliable and valid instrument (Custers et al 2002a, 2002b,

Wassenberg-Severijnen et al 2003).

Analysis of the contents of COPCA and TIP

At four and six months CA, we made video recordings of an intervention session. For 43 infants,

video recordings of infant physical therapy sessions were available. The remaining three infants

(all TIP) had too few intervention sessions to make a video recording. The contents of the sessions

were analyzed with a standardized observation protocol that we recently developed for analysis of

physiotherapeutic intervention sessions in young infants (Blauw-Hospers et al 2010). The protocol

is based on knowledge about infant physical therapy and analysis of directly observable physio-

therapeutic (PT) and caregiver actions. Inter- and intrarater agreement proved to be satisfactory

(Blauw-Hospers et al 2010). The observation protocol classifies PT actions into eight main catego-

ries: 1) Family involvement and educational actions; 2) Communication; 3) Facilitation; 4) Sensory

experience; 5) Passive motor experience; 6) Self-produced motor behavior with no interference; 7)

Challenge to self-produce motor behavior where the infant is allowed and challenged to continue

activity; 8) Challenge to self-produce motor behavior, flowing over into therapeutic handling. We

added a variable which was the amount of postural support provided during PT actions.

The analysis was carried out with the help of the computer program “The Observer” (Noldus).

The Observer software is a tool for collecting and analyzing observational data in a descriptive

and quantitative way. The outcome parameters of the analysis were the relative amounts of time

spent on PT actions. The category self-produced motor behavior without interference of the

physical therapist or the caregiver that is present in the original protocol was left out of the cur-

rent analyses because of a qualitative difference between the two groups during the periods of

entirely spontaneous activity (Dirks et al, submitted for publication). Previously, we reported that

PT actions at four months were largely comparable to those at six months (Dirks et al, submitted

for publication). Assuming that the two measurements at four and six months represented the ac-

tions during the intervention period better than a single measurement, we used the average of the

four-month and six-month values for PT actions in the correlations with developmental outcome.

Data-analysis

Statistical analyses were performed using the computer package SPSS (version 14.0). To evaluate

the effect of type of intervention (COPCA or TIP; group classification on the basis of the video

contents of the physical therapy session) on developmental outcome at 6 and 18 months CA, the

non-parametric Mann-Whitney test was used since the data were not normally distributed. Dif-

ferences having a p-value <0.05 were considered statistically significant.

To examine associations between the contents of intervention and developmental outcome,

univariate analyses were applied first. To minimize the possibility that associations could be

explained by the influence of potential confounders, such as the infant’s baseline condition (neu-

rological outcome and AIMS score at three months, the presence of a severe brain lesion), social

factors (maternal education, being the first child or not), and factors related to the intensity of

physical therapy treatment (number and duration of sessions), we applied multivariate statistics.

Linear regression techniques were used, since the data were skewed to a minimal extent only. In

this way the contribution of the relative duration of PT actions to outcomes on the NOS, AIMS,

BSID-II, and PEDI was assessed. For the associations between the contents of the intervention

and the AIMS score at 18 months CA, logistic regression analysis was carried out, given that at 18

months the AIMS suffered from a ceiling effect. Therefore, we dichotomized the AIMS score into

58 and <58. Correlations with a p-value ≤ 0.01 were considered statistically significant.

140 141

results

Developmental outcome according to randomized group allocation

The developmental outcome of the three infants in the TIP group who had too few intervention

sessions to make a video recording was similar to the other 22 infants who received TIP. There-

fore, we decided to include them in the RCT analysis.

The neurological condition at 6 and 18 months CA in the COPCA and TIP groups was similar

(Table 7.2). At six months, one infant in the COPCA group had a normal-suboptimal neurological

condition, 16 were classified as MND, and 4 showed an abnormal neurological condition. In the

TIP group, 20 infants were classified as MND and 5 had an abnormal neurological condition. At

18 months CA, ten infants, five in each group, had developed CP. One infant in the COPCA group

and two infants in the TIP group were neurologically normal. The remaining infants (15 COPCA,

16 TIP) had complex MND. Two infants in the TIP group did not return to the follow-up at 18

months. The NOS of the COPCA group at 18 months tended to be a bit better than the TIP group

(COPCA median value 31 versus TIP median value 27: Table 7.2) but the difference did not reach

statistical significance.

Performance on the AIMS at 6 and 18 months CA was identical for both groups. This was true

for the total AIMS scores and the scores on the subscales (Table 7.2). The data revealed that at the

age of 18 months the AIMS suffered from a ceiling effect (Darrah et al 1998, Liao and Campbell

2004). It only could differentiate between children with and without CP.

The MDI scores at 6 and 18 months CA did not differ for COPCA and TIP. At six months CA,

the median value of the MDI in the COPCA group was 106.5 and in the TIP group 115.5; at 18

months CA, the median values of the MDI were 100 and 98, respectively. The data also indicated

that in both groups the MDI decreased between 6 and 18 months CA. The decrease in MDI score

from 6 to 18 months CA was statistically significant in the TIP group (p=0.001; Figure 7.1) but

non-significant in the COPCA group (p=0.07). The change in MDI score between 6 and 18 months

CA was affected by the level of maternal education (p=0.01) and the type of intervention (p=0.03).

The data indicated that especially those infants in the COPCA group whose mother had a lower

level of education showed the smallest drop in MDI score (Figure 7.2). At 18 months, there were no

differences between COPCA and TIP on the PEDI (Table 7.2).

6 months Ca COPCa (n=21) tIP (n=25)

TINE (n=46) Normal-suboptimal MND Abnormal

1164

0205

AIMS (median value; range) 18 (6-22) 17 (9-22)BSID-MDI (median value; range) 106.5 (50-135) 115.5 (84-145)

18 months Ca COPCa (n=21) tIP (n=23)

Hempel (n=44) Normal Complex MND Abnormal / CP

1155

2165

NOS (median value; range) 31 (9-51) 27 (10-47)AIMS (median value; range) 57 (10-58) 58 (16-58)BSID-MDI (median value; range) 100 (50-119) 98 (72-135)PEDI – Functional skills scale(median value; range)Selfcare MobilitySocial

24 (13-38)35 (6-44)21 (11-36)

24 (11-27)29 (6-37)22 (11-31)

AIMS = Alberta Infant Motor Scale, BSID-MDI = Bayley Scales of Infant Development - mental developmental

index, CA = corrected age, CP = cerebral palsy, MND = minor neurological dysfunction, NOS = neurological

optimality score, PEDI = Pediatric Evaluation of Disability Inventory, TINE = Touwen Infant Neurological

Examination

table 7.2. Developmental outcome on group level (randomized groups).

142 143

low middle high

6 mo 6 mo18 mo 18 mo

ns **

140

120

100

80

60

40,00

20,00

0,00

-20,00

-40,00

Maternal Education

dete

rior

atio

n im

prov

emen

t

COPCA(n=21)

TIP(n=23)

tIP

COPCa

BSI

D -

MD

IB

SID

- M

DI

Figure 1: Bayley Scales of Infant Develop-ment: MDI scores at 6 and 18 months in the COPCA and TIP group. Vertical bars, range; horizontal lines, median values; boxes, interquartile range; ns, not significant. ** p=0.001 (Wilcoxon signed ranks test).

Figure 2: Relative deterioration in MDI score between 6 and 18 months and maternal education. The graphs illustrate that MDI is affected by maternal education (p=0.01) and type of intervention (p=0.03) (MANOVA).

Associations between treatment principles and developmental outcome

The results of the RCT showed only minimal differences between COPCA and TIP. We did,

however, observe changes in developmental outcome. To examine whether these changes could be

associated with treatment principles used by the physical therapist or caregiver during interven-

tion or with confounding factors, we applied process evaluation to the data. First, we analyzed

the contents of the intervention sessions. The details of the analysis are described in Dirks et al.

(submitted for publication). Typical COPCA components were: 1) physical therapist coaches the

parent (coaching model); 2) to stimulate self-produced motor behavior at the limit of an infant’s

capabilities; and 3) infant is challenged to produce motor behavior by him/herself and is allowed

to continue activity. Typical TIP components were: 1) physical therapist teaches the infant and

trains the parent (teacher-learner model); 2) in handling techniques; 3) in sensory stimulation; and

4) to challenge for self-produced motor behavior that is followed by a handling technique.

Preliminary analyses showed that the associations between PT actions and developmental

outcome measurements differed for infants with and without CP. An overview of the associations

between treatment principles and developmental outcome is presented in Table 7.3.

In infants who developed CP (n=10) no statistically significant associations were found be-

tween specific PT actions and developmental outcome at six months CA. Some PT actions were

correlated with outcome at 18 months in the infants with CP. The multivariate analyses showed

that more time spent on family involvement and educational actions, more time spent with

postural support at the limit of the infant’s capabilities, and more time during which the infant

was challenged in a widely varying way to produce motor behavior and was allowed to continue

this activity were associated with a higher score on the functional mobility scale of the PEDI at 18

months CA (all p<0.01; Table 7.3). More time spent with passive experience was associated with a

lower NOS at 18 months CA (p<0.01).

In the children without CP (n=33), the amount of time spent on sensory and passive experience

showed a positive correlation with the MDI at six months (both p<0.01), but these associations did

not persist through to the age of 18 months. In children without CP, more time spent on handling

techniques was associated with a lower score on the functional mobility scale of the PEDI at 18

months (p<0.01).

Allocation of intervention based on video analysis

Since the treatment application was very heterogeneous, we decided to regroup the infants on the

basis of the contents of the video into intervention subgroups. The infants who received COPCA

were allocated based on the contents of the intervention to COPCA++ or COPCA+. The interven-

tion sessions of infants who got TIP were classified as TIP++ or TIP+. The ++ notation indicates

that the intervention was performed fully according to the principles of COPCA or TIP. In the

case of TIP, this meant that the principles of the original concept of the Bobaths were used. The

+ notation indicated that the contents of the intervention was more diverse. For COPCA this

implied that COPCA principles were applied incompletely, while for TIP, that treatment consisted

144 145

a) Infants who developed CP (n=10) Outcome measure Model Beta CI p

Family involvement and educational actionsU PEDI - functional mobi-

lity scale 18 moFameduc 0.54 7.59-70.24 0.02

M PEDI - functional mobi-lity scale 18 mo

FameducAIMS 3

0.520.51

11.91-63.660.60-3.40

<0.01<0.01

Postural support - at the verge of infant’s capabilitiesU PEDI - functional mobi-

lity scale 18 moSupport verge 0.60 12.85-69.59 <0.01

M PEDI - functional mobi-lity scale 18 mo

Support vergeAIMS 3

0.560.48

15.09-62.240.56-3.24

<0.01<0.01

Infant challenged to produce motor behavior and allowed to continue activity (large variation offered)U PEDI - functional mobi-

lity scale 18 moCPMB - allowed large var 0.52 4.37-47.72 0.02

M PEDI - functional mobi-lity scale 18 mo

CPMB - allowed large var AIMS 3

0.550.56

10.41-44.570.83-3.53

<0.01<0.01

Passive motor experienceU NOS 18 mo Passive -0.61 -112.69- -22.91 <0.01M NOS 18 mo Passive

Frequency prior to 3 mo-0.790.46

-92.00- -54.720.05- -0.12

<0.01<0.01

b) Infants without CP (n=33)Outcome measure Model Beta CI p

Sensory experienceU MDI 6 Sensory 0.34 19.88-155.07 0.01M MDI 6 Sensory

AIMS 30.360.32

28.36-157.091.00-7.79

<0.010.01

Passive motor experienceU MDI 6 Passive 0.29 11.69-218.80 0.03M MDI 6 Passive

Neur exam 3 mo0.35-0.34

38.77-237.60-26.47- -3.91

<0.01<0.01

Facilitation techniquesU PEDI - functional mobi-

lity scale 18 moFacilitation -0.41 -23.86- -6.00 <0.01

M PEDI - functional mobi-lity scale 18 mo

FacilitationAIMS 3 mo

-0.360.25

-22.03- -4.290.02-2.69

<0.010.05

AIMS = Alberta Infant Motor Scale, CPMB - allowed large var = Infant challenged to produce motor behavior and allowed to continue activity (large variation offered), Fameduc = Family involvement and educational actions, Frequency = number of treatment sessions, MDI = Mental Developmental Index, M = multivariate, Neur exam = neurological examination, NOS = neurological optimality score, PEDI = Pediatric Evaluation of Disability Inventory, U = univariate

table 7.3. Overview of associations between PT-actions and developmental outcome at 6 and 18 months. Results of univariate and multivariate analysis.

of a mix of actions according to the original Bobath concepts along with current NDT principles

involving a more functional approach.

The classification COPCA++ versus COPCA+ was based upon the amount of time that

was spent on the typical COPCA actions where the physical therapist coaches the caregiver to

stimulate motor behavior at the limit of an infant’s capabilities and where the infant is chal-

lenged to self-produce motor behavior that is then continued by the infant. The distinction TIP+

versus TIP++ was made based on the time spent on training the caregiver, handling and pressure

techniques, sensory experience, a challenge to self-produce motor behavior followed by a handling

technique, and the amount of time that actions were performed while the pelvis of the infant in

supine position was lifted slightly by the hands of the physical therapist. Each infant was given a

score on the basis of the average time-values of PT actions observed during the two intervention

sessions on the basis of the percentile values of the present study. When the time spent on a PT ac-

tion was below the 33rd percentile, 1 point was given; a time score between the 33rd and 67th percen-

tile resulted in 2 points; and a time score over the 67th percentile was given 3 points. In this way a

total COPCA score (maximum: 3 actions * 3 points = 9) and a total TIP score (maximum: 5 actions

* 3 points = 15) were calculated. The cut-off for the distinction between the ++ and + classification

was the 67th percentile of the total score. This resulted in a new group allocation that is presented

in Table 7.4. The allocation of the groups based on the time spent on PT actions matched the clas-

sification of the second author (TD) on the basis of Gestalt perception of the video recording.

table 7.4. Reallocation into intervention groups.

Classification total group (n=43)

Infants with CP (n=10) Infants without CP (n=32)*

TIP ++ 14 3 11TIP + 8 2 5COPCA + 7 1 6COPCA ++ 14 4 10

*1 infant did not return to the follow up.TIP++ = classic NDT approachTIP+ = mix of classic NDT and a more functional approachCOPCA+ = COPCA principles applied incompletelyCOPCA++ = COPCA principles fully applied

146 147

Based on the new group allocation, we reanalyzed the outcome measurements. The groups did

not differ in neurological classification, and AIMS and MDI scores at 6 and 18 months. For the do-

main of mobility of the functional skills scale of the PEDI at 18 months, the infants who received

COPCA or TIP+ performed significantly better than the infants in the TIP++ group (Mann-Whit-

ney p < 0.01; Figure 7.3a). They also needed less caregiver assistance in the mobility domain at 18

months (p=0.03; Figure 7.3b). The differences in performance for both PEDI outcomes between

children who received COPCA or TIP+ and those who received TIP++ remained statistically sig-

nificant in the multivariate analyses (details not provided).

Figure 7.3a: Scores on the domain mobility of the functional skills scale of the PEDI at 18 months CA in the COPCA+, COPCA++, TIP+ and TIP++ groups. Vertical bars, range; horizontal lines, median values; boxes, interquartile range.* difference between TIP++ vs other three groups: p <0.01, a significance which persisted in the multivariate analysis.

Figure 7.3b: Scores on the caregiver assistance scale in the domain mobility of the PEDI at 18 months in the COPCA+, COPCA++, TIP+ and TIP++ groups. Vertical bars, range; horizontal lines, median values; boxes, interquartile range.**difference between TIP++ vs other three groups: p=0.03

COPCA++

*

* o

COPCA++COPCA+ COPCA+TIP+ TIP+TIP++ TIP++

50,00

40,00

30,00

20,00

10,00

0,00

* *30,00

25,00

20,00

15,00

10,00

5,00

0,00

3aPEDI - functional mobility scale

group allocation group allocation

3bPEDI - caregiver assistance scale (domain mobility)

21

21 3

Discussion

The RCT revealed that COPCA and TIP were associated with similar effects for developmental

outcome. But the process evaluation indicated that the virtual absence of difference in group

effect may partially have been caused by the extensive heterogeneity in intervention strategies

within the groups, especially within the TIP group. The process analysis indicated that there

was some evidence that important components of the COPCA intervention were associated with

an improved developmental outcome. The therapeutic components of intervention during early

infancy, which were associated with a better developmental outcome at 18 months, were family

involvement and educational actions, application of broad variations in challenging the infant to

produce motor behavior by him/herself and allowing the infant to continue this activity, and the

provision of postural support at the limit of an infant’s capabilities.

Methodological considerations

The major strength of this study was its double approach. The combination of the RCT with a

process evaluation made it possible to determine the influence of the intervention on developmen-

tal outcome. Another strength of this study was that the infants all received intervention during

the same age period, that is, from three to six months post-term. This made the COPCA and TIP

groups more comparable. There was hardly any attrition during the study. Only two infants did

not return to the follow-up at 18 months CA. The final strength was that we used a set of instru-

ments to document outcome, ranging from instruments measuring at impairment level (neuro-

logical examination) to instruments measuring the level of activity and participation (PEDI).

One of the limitations of this study was that we studied a relatively small group of 46 infants.

Although the infants were selected because they all showed definitely abnormal GMs at ten weeks

CA, they had a heterogeneous developmental outcome. Various studies indicated that the pres-

ence of definitely abnormal GM is associated with a high risk for developmental motor disorders

such as cerebral palsy (Prechtl 2001, Hadders-Algra 2004). In this study only a minority of the in-

fants was diagnosed with CP at 18 months. This limitation is inherent to developmental changes

in the central nervous system. Developmental outcome at 18 months CA was relatively good, since

the median values of the MDI were 98-100. It should be noted that the majority of infants showed

complex MND at 18 months, which puts the infant at risk for learning and behavioral disorders at

school age (Hadders-Algra 2005). Another limitation is that we used 18 months CA in this study

to evaluate the long-term outcome of the intervention. As mentioned above, some developmen-

tal problems do not emerge before school age. Therefore, we recommend that future research

should re-examine these children at school age. Finally, the major limitation of this study was the

heterogeneity of the composition of treatment in the TIP group. We anticipated this problem by

including a process analysis, thereby turning a major limitation into a major strength.

148 149

Physiotherapeutic considerations

The RCT revealed only minor differences between the COPCA and the TIP group. One possible

explanation for the small size of the effect is that intervention can affect developmental outcome

in children with brain dysfunction to a limited extent only (Ade-Hall and Moore 2000, Wasiak

et al 2004, Hoare et al 2007, Anttila et al 2008). Animal and human studies indicate that inter-

vention after a lesion of the brain at early age affects motor development considerably less than

cognitive development (Spittle et al 2007, Kolb and Gibb 2007). Another explanation for the lack

of effect could be the heterogeneity of the TIP treatment. For research purposes, it is quite night-

marish that treatment application, although based on the same theoretical background, can be so

diverse. In our opinion this heterogeneity is a major problem in the application of the RCT design

in pediatric physical therapy.

The RCT had two interesting results. First, there was an interaction effect between the type of

intervention and the level of maternal education in the relative drop in MDI score between 6 and

18 months CA. Infants whose mothers had a lower level of education showed less decline in MDI

score over time than infants whose mothers had completed higher education. This effect was es-

pecially present in the COPCA group. This suggests that mothers with lower education especially

benefit more from coaching by a therapist who uses COPCA principles, while mothers with higher

education are less affected by coaching as well as training performed by a therapist. Second, the

finding of a similar neurological outcome in the COPCA and TIP groups is intriguing considering

the diametrically opposed view of TIP and COPCA on the importance of the neurological para-

meters of muscle tone and atypical movements. In the TIP treatment, especially when applied to

the original concepts of Bobath and/or NDT, influencing tone by means of handling techniques

plays an important role in the activities of the therapist during intervention sessions. In COPCA

no attention is paid to these impairments. In other words, handling techniques aimed at influenc-

ing muscle tone and facilitating movement sequences to improve function do not seem to affect

neurological outcome.

Process evaluation indicated that specific components of the interventions in the study were

associated with an improvement in developmental outcome. We would like to stress that observed

associations are not causations. Associations between PT actions and outcome are unavoidably

contaminated by the child’s initial degree of impairment. An infant with a more serious impair-

ment elicits differences in the contents of PT actions than an infant with milder impairments

where the degree of initial impairment to a substantial extent determines later outcome. Our data

indeed revealed clear interactions between the infant’s condition and therapist actions. We there-

fore studied associations separately for infants with and without CP. In addition, we used multi-

variate analysis in order to be able to take into account the infant’s initial degree of impairment.

Nevertheless, it is also conceivable that other infant-specific factors which we did not include in the

analyses did affect the choice of PT actions and the child’s developmental outcome. Yet, the results

of the multivariate analysis may guide our thinking about the effectiveness of specific PT actions.

Several components of COPCA were associated with a higher score in the domain of mobility on

the functional skills scale of the PEDI. An example of this is the category of family involvement

and educational actions which in COPCA implies coaching of family members to develop their

own ways to care for the infant and to cope with the problems of the infant with special needs.

During the intervention, the coach listens, informs, and observes (hands-off), while the caregiver

is involved in daily routines with the child, including play, thereby creating a situation in which

caregivers feel free to explore and discuss alternative strategies. Second, wide variation in self-

produced motor activities (hands-off), trial-and-error experiences, and, if necessary, the provision

of minimal postural support creates a challenging environment in which the infant may explore

and practice his/her motor possibilities (Dirks et al, submitted for publication).

Some typical TIP actions were also associated with developmental outcome. The use of facili-

tation techniques such as handling, which are applied to reduce atypical functional activities and

to prepare the infant for optimally independent function (Dirks et al, submitted for publication),

were associated with a worse performance on the functional skills scale of the PEDI. On the other

hand, sensory and passive experiences were associated with a higher MDI immediately after the

intervention period. Perhaps sensory and passive experiences reflect situations of increased infant

attention which may promote cognitive development (Bahrick et al 2004). Our data indicated that

this beneficial effect disappeared over time.

Since the treatment application was very heterogeneous, we decided to regroup the infants on

the basis of the contents of the video into intervention subgroups. Although this approach lacks

the advantages of an RCT, it was interesting to see that infants in the COPCA and functional TIP

group scored better for domain of mobility on the functional skills scale and needed less car-

egiver assistance for domain of mobility on the PEDI than infants who received a TIP treatment

according to the original Bobath approach. This would indicate that the functionally oriented

interventions succeeded in influencing the performance of functional skills in a beneficial way.

It was interesting to observe that the effects of intervention were clearer at 18 months CA than

immediately after the intervention at 6 months CA. Perhaps this finding is related to the COPCA

approach which aims at supporting family members on the basis of an ongoing equal partnership

in order to uncover their own specific problem-solving strategies for caring and coping with the

infant with special needs.

Concluding remarks

The study showed that our nightmare had a good outcome. Extending the RCT with process

evaluation was necessary in order to answer the question of what components of intervention are

associated with developmental outcome. Our study showed that specific components of COPCA,

especially family involvement and educational actions, challenging the infant with a wide vari-

ation to produce motor behavior by him/herself and then to allow the infant to continue this

activity, and stimulating motor behavior at the limit of an infant’s capabilities according to the

principles of the NGST, were associated with improved developmental outcome.

150 151

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C H a P t E r 8

A ddI t Ion A l r e s u lt s

on o u tc om e m e A s u r e s

of t h e V I P P roj e c t

154 155

In this Chapter some of the results of questionnaires which were also assessed during the VIP

project, but were not the focus in the previous sections, are presented. Successively, attention is

paid to the Kent Infant Development Scales (KIDS), the Measures of Processes of Care (MPOC)

and the Nijmegen Questionnaire on Child-rearing Situations (NCSQ). Furthermore some prelimi-

nary results on postural control will be described. The results of the IMP, video recordings of ADL

activities and postural control will be published separately.

Kent Infant Development Scales

In total, 113 questionnaires (51 COPCA; 62 TIP) were returned (response rate of 82%). Especially at

3 months CA attrition was high (28%). At 6 and 18 months CA attrition was lower, but still ranged

from 8 to 19%. One possible explanation for the attrition rate was that despite the instructions

some caregivers did not complete the questionnaire before the next test session. Reasons they

mentioned were that they did not exactly know the corrected age of the infant, they didn’t see the

necessity to fill out the questionnaire at the right time, or they simply forgot. Caregivers with a

lower educational level or with known psychopathological problems tended to return the ques-

tionnaires less often.

The results showed that KIDS scores of infants in the COPCA group did not differ from those in

the TIP group. Both the total score and the scores on the five subscales were comparable at 3, 6, and

18 months CA (see Table 8.1). The possibility that under- or overestimation of the development of the

infant influenced the KIDS scores is present but almost negligible. The caregivers were only asked

to report whether they had noticed a specific behaviour, not to judge the development of the infant

(Stancin et al 1984).Some outliers were present in the data but they hardly influenced the results.

table 8.1. Median scores and ranges of the KIDS at 3, 6 and 18 months CA.

COPCa tIP3

(n=15)

6

(n=17)

18

(n=18)

3

(n=18)

6

(n=23)

18

(n=21)

Total 68.0

(62-122)

139

(106-189)

243.5

(214-252)

64.0

(31-118)

128.0

(77-221)

246.0

(177-252)

Cognition 16.0

(5-25)

33.5

(24-43)

50.5

(44-52)

15.5

(7-28)

31.0

(14-45)

50.0

(42-52)

Motor 16.0

(4-28)

37.5

(24-65)

77.0

(48-78)

15.0

(4-38)

33.0

(21-72)

75.0

(34-78)

Language 11.0

(5-18)

20.0

(9-24)

37.0

(30-38)

10.0

(3-17)

19.0

(11-34)

37.0

(29-38)

Self-help 8.0

(3-20)

23.5

(15-33)

38.0

(29-39)

6.5

(3-11)

21.0

(11-36)

38.0

(29-39)

Social 16.0

(8-27)

29.0

(22-37)

50.0

(44-51)

15.5

(8-25)

26.0

(13-44)

50.0

(38-51)

Measures of processes of Care (MPOC)

In total, 115 questionnaires (53 COPCA; 62 TIP) were returned (response rate of 83%). Caregivers

with a lower educational level or with known psychopathological problems tended to return the

questionnaires less often. From the 115 questionnaires only 81 (37 COPCA; 46 TIP) were valid

and could be used for the final analysis. The non-valid questionnaires contained a consider-

able amount of missing data, i.e. more than 29 items, resulting in too few answers to calculate a

domain score (Van Schie et al 2004). The large amount of non-valid questionnaires was mainly

caused by the frequent use of the category ‘not applicable’. At both 3 and 6 months CA eleven ques-

tionnaires were non-valid. At 18 months CA twelve questionnaires were non-valid. The caregivers

of the latter twelve infants reported that they had no experience with a rehabilitation setting or

hospital visit during the period between 6 and 18 months CA and judged the MPOC as not suitable

for their situation. They did however return the questionnaire, but answered most questions with

‘not applicable’.

Figure 8.1. Median values of COPCA and TIP on the five domains of the MPOC.

COPCA COPCA COPCATIP TIP TIP

7,00

6,00

5,00

4,00

3,00

2,00

1,00

0,00

7,00

6,00

5,00

4,00

3,00

2,00

1,00

0,00

7,00

6,00

5,00

4,00

3,00

2,00

1,00

0,00

3 3 36 6 618 18 18

7,00

6,00

5,00

4,00

3,00

2,00

1,00

0,00

7,00

6,00

5,00

4,00

3,00

2,00

1,00

0,00

Enabling and Partnership

General information

Coordinated and Comprehensive Care

Specific information

respectfulans Supportive Care

COPCA COPCATIP TIP

3 36 618 18

156 157

table 8.2. Median Scores and Ranges of the MPOC at 3, 6 and 18 months CA.

COPCa tIPmo n* median Range mo n* median Range

EP 3 13 4.3 3.9-6.6 EP 3 15 4.6 3.6-5.76 9 4.5 3.7-5.6 6 15 4.2 3.5-4.918 13 4.8 3.9-5.4 18 11 4.4 3.4-5.6

PGI 3 6

10 6

5.3 4.8

3.7-6.2 3.6-6.7

PGI 3 6

15 13

4.9 5.0

3.0-5.9 2.8-5.7

18 7 4.8 4.4-5.6 18 8 4.6 3.1-5.2PSI 3 13 5.1 4.1-6.2 PSI 3 14 4.7 3.7-6.4

6 8 5.3 3.7-6.0 6 17 4.5 2.9-5.618 11 4.9 3.5-5.7 18 11 4.5 3.5-5.1

CCC 3 6 18

13 7 12

4.7 5.2 4.8

4.2-6.7 3.9-5.5 3.7-5.5

CCC 3 6 18

13 16 11

4.7 4.6 4.3

3.5-5.4 3.5-5.4 3.6-6.1

RS 3 12 4.3 3.6-6.4 RS 3 15 4.3 3.2-5.76 10 4.4 3.7-6.1 6 16 4.2 3.6-5.018 14 4.4 3.4-6.3 18 10 3.7 3.2-5.6

CCC = coordinated and comprehensive care for child and family, EP = Enabling and partnership, mo = months corrected age, PGI = providing general information, PSI = providing specific information, RS = respectful and supportive care*n varies due to missing data on the specific domains

In accordance with the literature (van Schie et al 2004, Nijhuis et al 2007) we noted problems with

the MPOC caregiver ratings. Caregivers reported difficulties with judging the care provided by

different service providers. Often they had medical experiences with service providers in differ-

ent settings such as the University Medical Center and a regional hospital. Experience frequently

did not match. While participating in the VIP project they could also judge the attitude of the

physiotherapist. Therefore we recommend in line with Nijhuis et al (2007) that in future research

the family-centredness of different settings should be assessed separately.

Figure 8.1 and Table 8.2 represent the MPOC median scores and ranges. The median scores in-

dicated that caregivers in the COPCA intervention group in most domains are more satisfied with

the care they received than caregivers in the TIP group. Furthermore the satisfaction of caregivers

in the COPCA group with the care they received improved during the intervention period on the

domains enabling and partnership, coordinated and comprehensive care, and respectful and sup-

portive care. Caregivers in the TIP group became less content with the care they received during

the intervention period. Note however that none of the findings reached statistical significance.

Nijmegen Child-rearing Situation Questionnaire

In total, 111 questionnaires (52 COPCA, 59 TIP) were returned (response rate of 80%). Thirteen

percent of the caregivers indicated that they refused to fill out the questionnaire because of the

confronting nature of some of the questions. They were confronted with questions like “it would

have been better when my child had not survived” or “my child should be placed in a fosterhome”.

As explanation they mentioned the stress they perceived during the NICU stay whether their

child would survive or the loss of a twin brother or sister of their child. For this reason the NCSQ

probably is not the most suitable instrument to measure parental stress and the existing parenting

situation in a study group with high risk infants.

Only a few differences on subscales were observed between COPCA and TIP on the NCSQ

(Table 8.3). At 6 months caregivers in the COPCA group are more willingly to accept help regard-

ing educational matters. At 18 months caregivers in the TIP group are more optimistic about the

future perspective and they believe that their situation is more dependent on fate.

Postural control

Preliminary results on the first 20 infants of the VIP project showed that at 4 months CA infants

of the COPCA (n=9) and the TIP group (n=11) showed less direction-specific activity in supine and

sitting than age-matched typically developing infants. At the age of 6 months CA postural control

in the group of infants who had received COPCA intervention was in some aspects significantly

better than that of the group who had received TIP. Infants who received COPCA showed more

direction specific adjustments in sitting, more often recruited the complete pattern in supine and

sitting position and showed significantly less often a synchronous onset of postural muscle acti-

vity (de Graaf-Peters et al 2007). Postural performance at 6 months of the high risk infants who

had received COPCA intervention had improved to such an extent that it closely resembled that of

typically developing peers. This indicates that it is conceivable that the postural control of infants

at high risk for a developmental motor disorder may be influenced with the COPCA intervention.

Whether these results hold for the entire study group and whether they are still present at 18

months CA is currently being assessed.

Summarizing, the additional results show that the scores of the infants in the COPCA and TIP

group on the KIDS total score and subscores were comparable at 3, 6 and 18 months CA, findings

which are in line with the results which used the Mental Developmental Index of the Bayley Scales

of Infant Development as outcome parameter. Outcome in the various domains of the MPOC in-

dicated that there were no statistically significant differences between the COPCA and TIP group

at 3, 6 and 18 months CA. However, caregivers in the COPCA group tended to become more satis-

fied with the care they received during the study while the caregivers in the TIP group became

less content. The NCSQ showed some minor differences between the COPCA and TIP group, but

probably the questionnaire is not the most suitable instrument to measure parental stress and the

existing parenting situation in a study group with high risk infants due to the confronting nature

of some of the questions. Finally, preliminary data of the first half of the study group suggested

that COPCA presumably has a beneficial effect on postural control.

158 159

tabl

e 8.

3. M

edia

n sc

ores

and

ran

ges

of t

he N

CSQ

at 3

, 6 a

nd 18

mon

ths

CA

.

CO

PCa

tIP

3 (n=1

7)6 (n

=17)

18 (n=1

8)3 (n

=19)

6 (n=2

2)18 (n

=18)

a. S

ubje

ctiv

e Fa

mil

y St

ress

1.

3(1

.0-4

.0)

1.4

(1.0

-4.8

)1.

3(1

.0-4

.3)

1.3

(1.0

-3.3

)1.

3(1

.0-4

.3)

1.3

(1.0

-3.8

)

Acc

epta

nce

1.0

(1.0

-3.0

)1.

4(1

.0-3

.8)

1.0

(1.0

-4.3

)1.

0(1

.0-2

.3)

1.0

(1.0

-2.8

)1.

0(1

.0-3

.8)

Cop

ing

1.5

(1.0

-2.0

)1.

5(1

.0-2

.9)

1.4

(1.0

-2.4

)1.

4(1

.0-2

.3)

1.5

(1.0

-1.9

)1.

4(1

.0-2

.1)

Prob

lem

s1.

3(1

.0-2

.9)

1.4

(1.0

-3.7

)1.

5(1

.0-3

.1)

1.1

(1.0

-3.0

)1.

4(1

.0-2

.6)

1.7 (1.0

-2.9

)

Nee

d fo

r ch

ange

1.2

(1.0

-2.0

)1.

2(1

.0-3

.2)

1.2

(1.0

-2.7

)1.

2(1

.0-2

.0)

1.2

(1.0

-1.7

)1.

2(1

.0-1

.8)

Stra

in1.

6(1

.1-3

.6)

1.9

(1.0

-4.1

)1.

6(1

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)1.7 (1

.0-3

.3)

1.6

(1.0

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)1.

6(1

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.6)

Man

agin

g on

one

’s ow

n1.

5(1

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.0)

1.5

(1.0

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)1.

5(1

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.0)

1.8

(1.0

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)1.

5(1

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1.5

(1.0

-3.8

)

Plea

sure

1.0

(1.0

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)1.

2(1

.0-2

.4)

1.0

(1.0

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)1.

2(1

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1.0

(1.0

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)1.

1(1

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.0)

Rel

atio

n1.

4(1

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.0)

1.3

(1.0

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)1.

2(1

.0-2

.4)

1.4

(1.0

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)1.

2(1

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1.2

(1.0

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)

C. a

ttri

buti

ons

on t

he c

hild

rear

ing

situ

atio

n1.

8(1

.0-4

.0)

2.0

(1.0

-4.1

)1.

8(1

.0-4

.7)

2.0

(1.0

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)1.

8(1

.0-4

.2)

1.8

(1.0

-4.2

)

Eff

ort o

f sel

f and

par

tner

1.8

(1.1

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)2.

0(1

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1.8

(1.1

-2.8

)2.

0(1

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.1)

1.8

(1.1

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)1.

8(1

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lity

1.8

(1.0

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)2.

0(1

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1.8

(1.0

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0(1

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1.8

(1.0

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e of

par

tner

1.6

(1.2

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1.9

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6(1

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1.8

(1.2

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)1.

6(1

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.2)

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k di

fficu

lty

1.3

(1.0

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)1.

8(1

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.1)

1.2

(1.0

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)1.

4(1

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1.4

(1.0

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)1.

6(1

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.1)

Luck

/fat

e2.

2(1

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.0)

2.2

(1.0

-3.3

)1.7

*(1

.0-4

.7)

2.0

(1.0

-3.3

)2.

5(1

.0-4

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2.5*

(1.0

-4.2

)

D. E

xpec

tati

on fo

r he

lp1.

9(1

.0-5

.0)

2.3

(1.0

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)1.

6(1

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2.1

(1.0

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(1.0

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)

Sati

sfac

tion

4.5

(3.2

-5.0

)4.

2(3

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.0)

4.5

(3.8

-5.0

)4.

2(2

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.0)

4.5

(3.5

-5.0

)4.

3(3

.7-5

.0)

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d fo

r ch

ange

1.7 (1.0

-2.7

)1.

9(1

.2-3

.3)

1.7 (1.0

-3.0

)1.

8(1

.0-3

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1.8

(1.0

-2.5

)1.7 (1

.2-2

.3)

Nee

d fo

r he

lp1.

4(1

.0-3

.0)

1.8*

(1.0

-3.0

)1.

0(1

.0-3

.2)

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references

De Graaf-Peters VB, Blauw-Hospers CH, Dirks T, Bakker J, Bos AF, Hadders-Algra M. Development of postural control in typically developing children and children with cerebral palsy: possibilities for interven-tion? Neurosci Biobehav Rev 2007;31:1191-1200.

Nijhuis BJG, Reinders-Messelink HA, de Blecourt ACE, Hitters MWMCG, Groothoff JW, Nakken H, Postema K. Family-centred care in family-specific teams. Clin Rehabil 2007;21:660-671.

Stancin T, Reuter J, Dunn V. Validity of caregiver information on the developmental status of severely brain-damaged young children. Am J Ment Defic 1984;8:388-395.

Van Schie PEM, Siebes RC, Ketelaar M, Vermeer A. The Measure of Processes of Care (MPOC): valida-tion of the Dutch translation. Child Care Health Dev 2004;30:529-539.

C H a P t E r 9

g e n e r A l dI s c u s s Ion

162 163

The main aim of this thesis was to contribute to the body of knowledge about the effects of early

physiotherapeutic intervention for young infants who are considered to be at risk for a develop-

mental motor disorder. Therefore existing programmes on the effects of early physiotherapeutic

intervention on motor and cognitive development were reviewed and a new physiotherapeutic

intervention programme COPCA was evaluated. The evaluation of the COPCA programme not

only focused on the effects on developmental outcome but also included a process evaluation of

the contents of the COPCA programme. Section 9.1 discusses the implications of the main find-

ings of the studies in this thesis. Reflections about the VIP project are offered in section 9.2. This

discussion ends with some implications for physiotherapeutic practice and recommendations for

further research (section 9.3).

9.1 Implications of the main findings

9.1.1 The effect of early intervention on developmental outcome

The systematic reviews on the effects of existing early physiotherapeutic intervention program-

mes on motor (Chapter 3 and 4) and cognitive development (Chapter 4) of infants with a high risk

for developmental motor disorders aged less than 18 months corrected age (CA) resulted in three

important findings. First, the effect of early intervention programmes which focus primarily on

the improvement of motor development is inconclusive. It remains a matter of debate to what

extent early intervention may facilitate outcome in various developmental domains. General

developmental programmes seem most promising to influence motor and cognitive development.

Second, we recommend that the choice of an early intervention programme should be adapted to

the infant’s age. It turned out that the type of intervention which might be beneficial for infants

at preterm age differs from the type which is effective in infants who have reached at least term

age. At preterm age infants seem to benefit most from intervention which aims at mimicking the

intrauterine environment, such as NIDCAP intervention. After term age, general developmental

programmes in which parents learn how to promote infant development can exert a positive effect

on motor and cognitive development. This finding is consistent with the results of studies on the

effect of early intervention which primarily aim at improving cognitive outcome. Third, interven-

tion programmes according to the principles of NDT or Vojta do not show a beneficial effect on

motor or cognitive development in infants at high risk for developmental disorders in the first

postnatal years. A limitation of the systematic reviews was that the contents of the programmes

and the methodological quality differed too much to allow for a meta-analysis of the data. The

findings of our reviews are in line with similar reviews performed by others investigating the ef-

fect of early intervention (Simeonsson et al 1982, Parette and Hourcade 1984, Casto and Mastrop-

ieri 1986, Shonkoff and Hauser-Cram 1987, Parry 1992, Turnbull 1993, Guralnick 1997, Majnemer

1998, Butler and Darrah 2001, Spittle et al 2007).

One of the aims of the VIP project was to evaluate the effect of two early intervention pro-

grammes on developmental outcome. COPCA can be considered as an example of a general devel-

opmental programme while TIP, the traditional approach towards infant physical therapy in the

Netherlands, is based for an important part on the principles of NDT treatment. Overall we found

only minor differences in developmental outcome till the age of 18 months between the COPCA

and TIP group. A possible explanation for this small size of the effect is that intervention can af-

fect developmental outcome in children with brain dysfunction to a limited extent only (Ade-Hall

and Moore 2000, Wasiak et al 2004, Hoare et al 2007, Anttila et al 2008).

Developmental outcome of the infants in the VIP project was relatively good as the median

values of the MDI were 98-100 and only a minor part of the groups were diagnosed with CP at

the age of 18 months CA. It should be noted however that the majority of infants showed complex

MND at 18 months, which puts the infant at risk for learning and behavioural disorders at school

age (Hadders-Algra 2005). Important findings of the VIP project were: 1) Neurological outcome at

6 and 18 months in the COPCA and TIP group was similar. This was interesting considering the

position that muscle tone and abnormal movement patterns, which are two important aspects

of neurological condition, have in both interventions. In TIP normalization of muscle tone and

movement patterns plays an important role (Howle 2002), while in COPCA no attention is paid to

these impairments. In other words, therapeutic techniques aiming to influence muscle tone and

movement patterns, do not seem to affect neurological outcome. 2) Motor performance as mea-

sured with the AIMS was similar at 6 and 18 months CA. Where the preliminary analyses for the

first half of the study group showed that infants who received COPCA tended to perform better

on de sitting subscale of the AIMS (Chapter 4), this result could not be replicated when we ana-

lyzed the data of the entire study group (Chapter 7). 3) Although the overall cognitive outcome was

comparable between the COPCA and TIP group at 6 and 18 months CA, we observed a decrease

in the MDI score over time which was statistically significant in the TIP group. This decrease in

cognitive outcome over time is in line with findings in other studies (Vohr and Garcia Coll 1985,

Aylward 1989, Bhutta et al 2002, Ment et al 2003). The change in MDI score between 6 and 18

months CA was affected by the level of maternal education and the type of intervention. Infants

whose mother had a lower level of education showed less decline in MDI score over time than

infants whose mother had finished a higher education. This effect was in particular present in

the COPCA group. This suggests that in particular mothers with a lower education benefit more

from the suggestions made by a physical therapist that uses COPCA concepts, while mothers

with a higher education seem less affected by suggestions of a physical therapist. 4) At 18 months

CA there were no differences between COPCA and TIP on the PEDI. However, it turned out that

the score on the functional mobility scale of the PEDI was associated with several elements of the

intervention (see 9.1.2). 5) Results from the questionnaires (Chapter 8) indicated that caregivers in

the COPCA group tended to become more satisfied with the care they received during the study

while the caregivers in the TIP group became less content (MPOC). Moreover the caregivers in

the COPCA group were more willingly to accept help regarding educational matters (NCSQ). 6)

Data from the first half of the study group suggested that COPCA presumably has a beneficial

effect on postural control. Infants who received COPCA showed more direction specific adjust-

ments in sitting, more often recruited the complete pattern in supine and sitting position and

164 165

showed significantly less often a synchronous onset of postural muscle activity (de Graaf-Peters

et al 2007).

9.1.2. Evaluation of the application of COPCA in daily practice.

The application of COPCA in daily practice was evaluated by means of video analysis. This pro-

cess evaluation is a prerequisite for the implementation of COPCA within paediatric physiotherapy.

It is acknowledged that designing intervention programmes requires more fundamental knowl-

edge about the relations between function, activities and participation than is offered by RCT

research (May et al 2007, Hullegie 2009). Complex therapeutic interventions comprise a number

of separate elements which seem essential to the proper functioning of the interventions, which

may act both independently and interdependently. The components usually include behaviours,

parameters of behaviours (e.g. frequency, timing), and methods of organizing and delivering those

behaviours (e.g. type(s) of practitioner, setting and location) (Medical Research Council 2000).

Randomised controlled trials (RCTs) are considered to be the most rigorous way to evaluate

the effectiveness of interventions, regardless of their complexity. Because of their multifaceted

nature and dependence on social context, complex interventions pose methodological challenges,

and require adaptations to the standard design of such trials (Oakley et al 2006). Process evalua-

tion is needed to describe what really happens in an intervention and to explore hypotheses about

why and how interventions are effective or ineffective (Lettinga et al 2002, Flottorp et al 2003,

Oakley et al 2006). Process evaluation is especially necessary in trials, where the ‘same’ inter-

vention principles may be implemented, but applied and received in different ways. This thesis

contributes to these issues in two ways. First, by developing a standardized observation protocol

which allowed for an objective quantification of the contents of physical add therapy in infancy.

Second, by combining the RCT with the process evaluation it was actually possible to explore

whether specific aspects in the contents of intervention could be associated with developmental

outcome. The study described in Chapter 7 in which we extended the results of the RCT with

process evaluation is a good example of integrating the knowledge about the contents of interven-

tion with the scientific approach of a RCT. The process evaluation has helped deepen our under-

standing of what actually happens during the intervention and which PT-actions contribute to an

improved developmental outcome.

Our inventory has resulted in a standardized observation protocol detailing essential elements

and core features of physiotherapeutic intervention, which permitted analysis of the contents

of physical therapy interventions for infants (see Chapter 5). The protocol was proven to meet

important psychometric requirements and captured nearly all physiotherapeutic actions which

were performed during intervention sessions of infants at high risk. It is an instrument which may

be used to assess heterogeneity in physical therapy as it offers a tool to describe operationally what

therapists do within treatment sessions across varying frames of theoretical references. Four con-

clusions can be drawn from the three studies (Chapter 5-7) which applied the observation protocol.

First, traditional infant physical therapy is characterized by a large heterogeneity in implementa-

tion. Presumably this heterogeneity is brought about by the application of techniques belonging to

different treatment approaches. Not only actions belonging to the concept of Neurodevelopmental

Treatment (NDT) including baby treatment - approaches well known for their heterogeneity in

application (Bly 1991) - are incorporated into treatment but also actions from a more functional

point of view. Research demonstrated that eclectic approaches tend to have smaller effects due to

the fact that there are more confounding factors which might influence the effect size (Mandich et

al 2001).

Second, the analysis of the contents of physical therapy sessions indicated a discrepancy

between actual clinical practice and the physiotherapist’s interpretation of his or her own actions.

The evaluation of the video’s taught us that physical therapists often do other things than they

intend to do. This indicates that it is very important to have a uniform language within physi-

otherapeutic practice. Currently, different treatment approaches use the same word to describe

different things. For instance, let us focus on the word “active”. NDT treatment aims to give the

child sensorimotor experience through the child’s active responses to specialized handling (Brice,

Bobath Centre, instruction video 1990). This implies that the active behaviour of the child is in

part a reaction to the handling technique of the PT. In functional therapy and COPCA “active”

refers to self-produced activities of the child. These frames of reference may differ more often

than we realize, resulting in miscommunication. Video feedback might serve as a tool to capture

this type of misunderstanding and subsequently add to the uniformity in the language used by

therapists. This means that only the evaluation of real action and communication through stand-

ardized, computer-based video-analysis of treatment sessions will provide insight in the reality of

practice.

Third, analysis of the newly developed programme COPCA showed many differences in the

contents of COPCA and TIP (Chapter 6). The differences found between COPCA and TIP corres-

ponds to the theoretical frameworks of both approaches (see Table 6.1). They included differences

in the role of caregivers and the approach of families, the application of educational actions

towards the infant and in activities to stimulate the infant’s sensorimotor development. These

findings indicate that COPCA and TIP based on NDT differ largely justifying the label ‘novel

intervention’ for the COPCA programme.

Fourth, the process evaluation indicated that several aspects of the COPCA intervention were

associated with an improved developmental outcome. Infants seem to benefit the most from

activities that add to a challenging environment in which large variation, trial-and-error experi-

ences and minimal postural support is offered. The physical therapist can coach the caregivers to

incorporate these aspects into all daily activities. Some typical TIP actions were also associated

with developmental outcome. The use of facilitation techniques like handling, which are ap-

plied to reduce atypical functional activities and to prepare the infant for optimally independent

function (Dirks et al, submitted for publication), was associated with a worse performance on the

functional skills scale of the PEDI. The use of sensory and passive experience showed a positive

166 167

short term effect on the cognitive development of the infants. Although the results of the VIP

project seem promising, it is important to realize that the results of the study should be interpret-

ed with caution. After all, associations do not directly mean that there is a causal relation between

a PT-action and developmental outcome. Furthermore the associations between PT-actions and

outcome are unavoidably contaminated with the child’s initial degree of impairment. Multivariate

statistics can be applied to account for this problem but still more research is needed to examine

the influence of the contents of intervention on developmental outcome.

To summarize, although the differences between the COPCA and TIP group on several aspects of

developmental outcome on RCT level are small, the results suggest that COPCA is more promis-

ing than TIP to influence aspects of developmental outcome. This is reflected in the findings that

infants who received COPCA decline less in cognitive function over time and that COPCA pos-

sibly has a beneficial influence on postural control. The most important argument that points to

a promising effect of COPCA is that key-elements of COPCA, i.e. coaching, variation, challenging,

and exploration, are associated with an improved developmental outcome on the functional mo-

bility scale of the PEDI. Hence combining the RCT with a process evaluation turned out to be the

ultimate combination to examine the research questions. The possible promising effect of COPCA

is in line with the results from the systematic reviews which indicated that general developmental

programmes are the most promising to influence developmental outcome in infants with or at

high risk for developmental motor disorders.

9.2 the VIP project; strengths and limitations

The early detection of infants at high risk for developmental motor disorders, such as cerebral

palsy (CP) offers the opportunity for intervention at young age. Research showed that the best

time window for early intervention in terms of active stimulation of the infant lies between 40-44

weeks post menstrual age and 15 months postnatally (De Graaf-Peters and Hadders-Algra 2006).

In the VIP project the infants received intervention from three to six months which lies entirely

within this recommended time window.

Selection procedure and study group

The infants were selected to participate in the VIP project when they showed definitely abnormal

general movements at the age of ten weeks corrected age. Various studies indicated that the pre-

sence of definitely abnormal GM is associated with a high risk for developmental motor disorders

(Prechtl 2001, Hadders-Algra 2004). It turned out that approximately 25% of the infants who had

been admitted to the NICU showed definitely abnormal General Movements (GMs). The response

to participate in the VIP project was 75% which resulted in a final study group of 46 infants. Chief

among the reasons for families to refuse participation were the distance to the UMCG and the

load and duration of the study.

One of the limitations of the VIP project was that we studied a relatively small group of 46

infants. Although power analysis before the start of the project showed that a sample size of 40

infants would be appropriate to detect differences in treatment, it is still a very small group to

detect real, and clinically worthwhile, differences in treatment. This is even more complicated by

the fact that is considered unethical to assign participants to a non-treatment control group (Maj-

nemer 1998, Siebes et al 2002). Also in the VIP project we encountered this challenge. Therefore it

was only possible to compare the effects of COPCA and TIP. The differences which we observed in

the RCT represent only the additional value of COPCA compared to TIP. It would be interesting

to know what the effects of both COPCA and TIP would be when they are compared to a group

that received no intervention. These two aspects indicate that we have to be very careful with the

interpretation and generalization of the results of the VIP project.

Another issue, which poses a challenge to the possibility to detect differences in develop-

mental outcome between two groups of high risk infants, is that development in early childhood

is very variable. In young infants brain dysfunction is expressed as generalized and unspecific.

Infants with a left-sided intraventricular hemorrhage may respond with signs of hypotonia,

hypertonia, hypokinesia or with a hyperexcitability syndrome (Hadders-Algra 2004). To make it

even more complicated, brain dysfunction present at birth is not always permanent. Due to neu-

rodevelopmental changes in the brain part of the infants with neonatal neurological symptoms

may recover (Hadders-Algra 2002, 2004). Note that also the opposite is possible (Hadders-Algra

2002, 2004). This makes the developmental course of infants with a high risk for developmental

motor disorders a fairly unpredictable process (Hadders-Algra 2002, Lebeer and Rijke 2003).

Evaluation and outcome measures

One of the strengths of the VIP project was that we studied the effect of intervention on short

term (immediately after the intervention at 6 months CA) and on long term (one year after the

intervention at 18 months CA). The outcome measures used in the VIP project to evaluate the

developmental outcome were age specific and belong to the international standard. It is however

acknowledged that standardized outcome measures which are often applied to measure changes

in developmental outcome are in general characterized by a lack of sensitivity to detect small

changes in motor or cognitive development although these small changes may have an important

influence on the functional abilities of the child. The use of discriminative measures, which focus

on the comparison of a child’s score with an age-equivalent score, may be one of the reasons that

so little effect of early intervention is found (Ketelaar et al 1998).

Neuromotor outcome was evaluated with two age specific instruments; the TINE (Touwen

Infant Neurological Examination; Hadders-Algra et al 2010) from three to six months and the

Hempel assessment at 18 months CA (Hempel 1993). Additionally the Neurological Optimality

Score (NOS; Huisman et al 1995) was used which is known to be an excellent instrument to evalu-

ate subtle differences in neurodevelopmental outcome (Bouwstra et al 2006). A striking finding

was that the neurological outcome in the COPCA and TIP group were similar at 6 and 18 months.

Considering the opposite view of COPCA and TIP on two important aspects of neurological

168 169

condition, i.e. muscle tone and abnormal movement patterns this was an interesting finding. In

COPCA no attention is paid to these impairments while in TIP treatment, especially in the classic

NDT variant, they play a fundamental role. It seems that therapeutic techniques aiming to influ-

ence muscle tone and movement patterns applied during early infancy do not affect neurological

outcome at 18 months.

While only a minority of the infants at high risk for developmental motor disorders develops

cerebral palsy (Marlow 2004), more than 50 percent shows minor neurological dysfunctions

(Stahlmann et al 2007). Although neurological outcome did not differ between both groups,

also the majority of the infants who participated in the VIP project (n=31) had complex minor

neurological dysfunction (MND) at 18 months CA. During the intervention period most infants

continued to show signs of neurological dysfunction, indicating that they were still in need of

physiotherapeutic guidance. The presence of complex MND during early infancy is a risk-factor

for the development of MND, DCD, attention and behavioural problems at school age (Jongmans

et al 1997, Hadders-Algra 2003, 2004, Groen et al 2005).

Motor development in the VIP project was evaluated with the AIMS. One problem was that at

18 months the AIMS suffered from a ceiling effect as the majority of the infants already reached

the stage of independent walking. Therefore it was not possible to differentiate between the

motor outcome of the COPCA and TIP group at the follow-up. Another problem associated with

the AIMS is that it mainly focuses on the observation of milestones and specific aspects of motor

behaviour. What we need is a reliable, valid and sensitive instrument which in addition to mo-

tor milestones pays attention to qualitative aspects of motor development. Possibly the recently

developed Infant Motor Profile (IMP; Heineman et al 2008) can solve this problem. The IMP is a

video-based assessment of motor behaviour in infancy that does not only address the infant’s mo-

tor abilities but also addresses variation in motor behaviour, the ability to select movement strate-

gies, symmetry, and fluency of motor behaviour. Currently the data of the IMP for the infants who

participated in the VIP project are being processed.

Cognitive outcome was assessed with the mental scale of the Bayley Scales of Infant Develop-

ment (BSID). The BSID mental scale, like most developmental instruments which measure cogni-

tive development in young children, heavily relies on fine motor skills. This will result in lower

scores for motor-impaired infants. Recently an adapted Low Motor version has been developed

especially for children with motor impairments in which the mental scale is less dependent of the

motor capacities of the child. Ruiter (2007) showed that the children with motor impairments

scored significantly higher on the Low motor version compared to the standard version of the

BSID mental scale. Probably this version gives a more adequate representation of the cognitive

abilities of infants with developmental motor disorders and can be used in future research.

One should keep in mind that the questionnaires we used, i.e. the KIDS, MPOC, NCSQ,

carry the risk that caregivers reflect on desired rather than actual behaviour. It is furthermore

important to realize what the effect might be of certain questions on the caregivers. For instance

the NCSQ contains many confronting questions for parents who had experienced stressful

times when the infant was at the neonatal intensive care unit. By completing the MPOC many

caregivers reported difficulties with judging the care provided by different service providers. Often

they had not matching medical experiences with service providers in different settings such as

the University Medical Center and a regional hospital. While participating in the VIP project they

could also judge the attitude of the physiotherapist. Therefore we recommend in line with Nijhuis

et al (2007) that in future research the family-centredness of different settings should be assessed

separately.

Intervention

Research into the evaluation of the effectiveness of early intervention is very complex for a host of

reasons. Chief among them is that physical therapy is not a specific treatment that is delivered in a

standardized manner. In other words, there is no discrete dosage administered under specific, in-

variable procedures in conditions that are held constant. The frequency or duration could be held

constant, but the procedures depend upon the therapists’ skill level and specific aims and vary

accordingly. While the treatment setting could be standardized, the child’s family or the child’s

responses to the treatment could never be (Butler and Darrah 2001).

Some infants already got physical therapy before they entered the VIP project and since they

- as already mentioned - continued to show signs of neurological dysfunction during the interven-

tion period most infants also received some physical therapy sessions after the age of 6 months.

After the intervention period the suggestions of the paediatrician regarding physical therapy were

followed. Twenty-five of the infants (7 COPCA, 16 TIP) received more than 10 sessions after the

age of 6 months, including the ten infants who developed CP. To cope with this additional hetero-

geneity in therapy the frequency of additional intervention sessions was taken into account in the

multivariate analyses of outcome.

It turned out that TIP was characterized by a large heterogeneity in treatment application

which was inherent to the evolvement of treatment techniques and theoretical assumptions

over the years and to the incorporation of new treatment approaches into older ones (Bly 1991,

Howle 2002, Blauw-Hospers et al 2009). This heterogeneity makes it very difficult to interpret the

results because the contents of the individual treatments differ so much that the aspects which

are responsible for the observed changes cannot be distinguished. The process evaluation gave

us a reliable quantification of TIP in the Netherlands. It enabled us to tackle the problem of the

heterogeneity and answer the question which aspects of the TIP treatment are associated with

developmental outcome (Chapter 7).

The fact that most associations between PT-actions, in particular the typical COPCA ac-

tions, during the intervention period were associated with developmental outcome measures at

18 months CA is an interesting finding. Improvement of outcome can likely be expected when

a behavioural change has occurred. In the field of early intervention this means that interven-

tion strategies should be incorporated into daily caregiving tasks, which is one of the goals of the

COPCA programme (Dirks and Hadders-Algra 2003, Dirks et al submitted).

170 171

9.3 Implications for physiotherapeutic practice and directions for future research

Paediatric physical therapy for infants at high risk for a developmental motor disorder can be

considered as a field in development. No evidence is available that traditional therapy approaches,

such as NeuroDevelopmental Treatment and treatment according to Vojta have a positive effect

on the development of infants at high risk for developmental motor disorders (Blauw-Hospers

and Hadders-Algra 2005, Blauw-Hospers et al 2007, Spittle et al 2007). This is the reason that

Damiano wrote ‘Based on the existing evidence the traditional approaches demand serious recon-

sideration by those who still advocate them’ (Damiano 2006). The findings that evidence for the

effectiveness of the traditional EI programmes is limited initiated important shifts in the applica-

tion of therapy. The most important advances are emphasis on functionality instead of normal-

ity, the infant is an active participant of the therapy, and focus on family dynamics. Currently,

evidence is accumulating that intervention from a more functional approach focusing on general

development is most promising to influence developmental outcome in a beneficial way (Chapter

3 and 4). Research has indicated that the involvement of caregivers can be associated with positive

effects both on the development of the child (Ketelaar et al 1998, Law et al 1998, King et al 2004)

as well as on the caregivers in terms of an increase in parental skills, improved well-being and a

decreased level of parental stress (King et al 1996, Rosenbaum et al 1998, Jansen et al 2003, King et

al 2004, Raina et al 2005). Another factor which may add to the possibility that intervention will

sort a long term effect is the implementation of intervention strategies into daily activities. In the

COPCA intervention focus is on family function, family autonomy and activities and participation

in daily life activities. The results of the VIP project suggest that this strategy may be effective.

In this thesis the first results on the implementation of the COPCA programme are described.

Although the effects on RCT level were small, the results from the process evaluation showed

that key aspects of COPCA, i.e. family involvement and educational actions, application of large

variation in challenging the infant to produce motor behavior by him/herself and allow the infant

to continue this activity, and stimulation of motor behavior at the verge of the infant’s capabili-

ties, were associated with improvements in developmental outcome at 18 months (Chapter 7).

To establish real insight in the effects and working mechanisms of COPCA on developmental

outcome more research has to be performed. It is likely that the effect of intervention is a combi-

nation of several factors which are related to the condition of the infant, the family, the therapist,

the contents of the intervention, and environmental factors. As some problems related to early

neurological dysfunction become more visible at school age future studies should include a longer

follow-up.

We described a first step in objectifying physiotherapeutic practice. By uncovering the

contents of infant physical therapy in the Netherlands we obtained insight into which actions

constitute practice and we were able to study relations with developmental outcome. A next step

in objectifying physiotherapeutic practice might be a study on the similarities and differences in

the application of infant physical therapy in other countries. Additionally more research must be

performed to optimize the psychometric requirements of the observation protocol.

Future research should not only focus on the development of new intervention strategies. Ade-

quate assessment techniques are also indispensable for the evaluation of the effectiveness of

intervention. There is a need for more scientific sound qualitative instruments which are able to

assess and evaluate the developmental outcome of infants at high risk for developmental motor

disorders more accurately. Current advances show that the psychometric properties of several

qualitative instruments are being examined (for instance the Infant Motor Profile (Heineman et

al 2008), the Touwen Infant Neurological Examination (Hadders-Algra et al 2010), and the Low

Motor Version of the Bayley Scales of Infant Development (Ruiter 2007)) suggesting that these

methods can be applied in future studies.

The implementation and evaluation of a new outcome measure or intervention programme into

clinical practice takes several years (Ketelaar et al 2008). Here a possible threat comes up. As

physiotherapists tend to include only techniques they perceive as helpful into their treatment, the

risk exists that a new intervention is not implemented in the way it was intended. Good education

is essential to diminish this threat. Although the results are still premature and more research

has to be performed, COPCA - a family friendly approach to intervention - has the potential to

influence developmental outcome of infants with high risk for developmental motor disorders in

a beneficial way. Currently another trial on the effectiveness of COPCA is carried out: Learn to

Move 0-2 years (L2M0-2). L2M0-2 is part of a multicenter trial on the effectiveness of intervention

in children and adolescents with CP. In L2M0-2 many of the limitations encountered in the VIP

project are accounted for. In the near future this will result in more knowledge about the effect of

early intervention on the developmental outcome of infants at high risk for developmental motor

disorders.

172 173

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S U M M a ry

Young infants, who are at risk for developmental motor disorders, such as cerebral palsy (CP) or

developmental coordination disorder, are often referred to early intervention services. The field of

early intervention consists of multidisciplinary developmental, educational and therapeutic

services provided to children from birth to 5 years of age. In this thesis early intervention is

restricted to paediatric physical therapy for infants up to 18 months corrected age (CA). The intro-

duction (Chapter 1) starts with an overview of the main influences on paediatric physical therapy

in the Netherlands. Paediatric physical therapy for infants at high risk for a developmental motor

disorder can be considered as a field in development. Based on findings that evidence for the

effectiveness of the traditional early intervention programmes (NeuroDevelopmental Treatment,

treatment according to Vojta, and Sensory Integration Therapy) is limited, shifts in the applica-

tion of therapy were initiated. The most important advances are a focus on functionality instead

of normality, the infant as an active participant of the therapy and the inclusion of the family.

The first aim of this thesis was to review the effects of early physiotherapeutic intervention on

motor and cognitive development of infants with a high risk for developmental motor disorders

aged less than 18 months (Chapter 3 and 4). The reviews showed that the effect of early intervention

programmes which focus primarily on the improvement of motor development is inconclusive.

It turned out that the type of intervention which might be beneficial for infants at preterm age

differs from the type which is effective for infants who have reached at least term age. At preterm

age infants seem to benefit most from intervention which aims at mimicking the intrauterine

environment. After term age, general developmental programmes in which parents learn how to

promote infant development can exert a positive effect on motor and cognitive development.

Traditional therapy approaches, such as NeuroDevelopmental Treatment and treatment according

to Vojta, do not influence the developmental outcome of infants at high risk for developmental

motor disorders.

The results of both reviews indicated that intervention programmes with an onset after term

age which focus on the stimulation of self-produced motor behaviour may exert a beneficial effect

on motor and cognitive development. This knowledge served as one of bases for the development

of a new type of physiotherapeutic intervention, COPCA. COPCA is the abbreviation of Coping

with and Caring for infants with special needs – a family centred programme. It is a family

relationship oriented programme consisting of two theoretical constructs. The first component

is formed by a family involvement and educational parenting component. The second theoretical

component, a neurodevelopmental one, is based on the principles of the Neuronal Group Selec-

tion Theory.

The second aim of this thesis was to evaluate the effects of the COPCA intervention on develop-

mental outcome. In the early intervention project VIP (in Dutch “vroegtijdig interventie project”;

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Chapter 2) 46 high-risk infants were randomly assigned to receive COPCA (n=21) or traditional

infant physical therapy (TIP; n=25) between 3 to 6 months CA. Developmental status of the infants

was assessed at 3, 4, 5, 6 and 18 months CA with instruments measuring at impairment level (neu-

rological examination) to instruments on the level of activity and participation (PEDI).

The randomized controlled trial (RCT) revealed that developmental outcome in both groups

was largely identical (Chapter 7), except for cognitive outcome. Ten infants were diagnosed with

CP at the age of 18 months CA. The majority of infants (15 COPCA, 16 TIP) showed complex minor

neurological dysfunction, which puts the infant at risk for learning and behavioural disorders at

school age. The RCT had two interesting results. First, there was an interaction effect between

the type of intervention and the level of maternal education in the relative drop in MDI score

between 6 and 18 months CA. Infants whose mother had a lower level of education declined less

in MDI score over time compared to infants whose mother had finished a higher education. This

effect was in particular present in the COPCA group. This suggests that in particular mothers

with a lower education benefit more from coaching by a therapist who uses COPCA principles,

while mothers with a higher education are less affected by coaching as well as training performed

by a therapist. Second, the finding of a similar neurological outcome in the COPCA and TIP group

is intriguing considering the opposed view of TIP and COPCA on the importance of the neuro-

logical parameters muscle tone and atypical movements. In the TIP treatment, especially when

applied to the original concepts of Bobath and/or NDT, tone influencing by means of handling

techniques plays an important role in the activities of the therapist during intervention sessions.

In COPCA no attention is paid to these impairments. In other words, handling techniques aiming

to influence muscle tone and facilitating movement sequences to improve function, do not seem

to affect neurological outcome.

Some supplementary findings of the VIP project are discussed in Chapter 8. Other data of the

VIP project are currently analysed (motor development in terms of the Infant Motor Profile and

postural control, and video-recordings of daily life activities).

The third aim of this thesis was to evaluate the application of COPCA in daily practice by means

of video analysis. This process evaluation is a prerequisite for the implementation of COPCA

within paediatric physiotherapy. The first step to achieve this was the development of a standard-

ized, theory based, observation protocol detailing essential elements and core features of paedi-

atric physiotherapeutic intervention, which permitted analysis of the content of physical therapy

interventions for infants (Chapter 5). The observation protocol classifies PT actions into eight

mutually exclusive categories: A) Family involvement and educational actions, B) Communication

, C) Facilitation techniques, D) Sensory experience, E) Passive motor experience, F) Self-produced

motor behaviour, no interference with PT /caregiver, G) Challenge to self-produce motor behav-

iour by him/herself; infant is allowed to continue activity, H) Challenge to self-produce motor

behaviour by him/herself; activity flows over into therapeutic handling. The protocol was proven

to meet important psychometric requirements and captured nearly all physiotherapeutic actions

which were performed during intervention sessions of infants at high risk for a developmental

motor disorder. The observation protocol may be used to assess heterogeneity in physical therapy

as it offers a tool to describe operationally what therapists do within treatment sessions across

varying frames of theoretical references. Four conclusions can be drawn from the three studies

(Chapter 5-7) which applied the observation protocol:

1) Traditional infant physical therapy is characterized by a large heterogeneity in implementation

which probably is caused by the application of techniques belonging to different treatment ap-

proaches.

2) A discrepancy exists between actual clinical practice and the physiotherapist’s interpretation of

his or her own actions.

3) The contents of COPCA and TIP sessions differed largely, suggesting that it is possible to

implement COPCA in daily practice by physical therapists who received special training in the

COPCA programme (Chapter 6). The differences found between COPCA and TIP corresponds

to the theoretical frameworks of both approaches. They included differences in the role of care-

givers and the approach of families, in the application of educational actions towards the infant

and in activities to stimulate the infant’s sensorimotor development.

4) Several aspects of the COPCA intervention, i.e. family involvement and educational actions, ap-

plication of large variation in challenging the infant to produce motor behaviour by him/herself

and allow the infant to continue this activity, and stimulation of motor behaviour at the verge

of the infant’s capabilities, were associated with an improved developmental outcome. The use

of facilitation techniques like handling was associated with a worse performance on the func-

tional skills scale of the PEDI. The use of sensory and passive experience was associated with a

short term positive effect on cognitive development.

It is important to realize that the results of this study should be interpreted with caution as the

observed associations do not automatically mean that a causal relation between contents and out-

come exists. Associations between PT actions and outcome are unavoidably contaminated with

the child’s initial degree of impairment. Multivariate statistics were applied to account for this

problem but still more research is needed to examine the influence of the contents of intervention

on developmental outcome.

In Chapter 9 the main findings of this thesis are summarised and discussed and placed into

perspective. Although the differences between the COPCA and TIP group on several aspects of

developmental outcome on RCT level are small, the results suggest that COPCA - a family friendly

approach to intervention - has the potential to influence developmental outcome of infants with

high risk for developmental motor disorders in a beneficial way. The most important argument

that points to a promising effect of COPCA is that key elements of COPCA, i.e. coaching, vari-

ation, challenging, and exploration, are associated with an improved developmental outcome.

Hence combining the RCT with a process evaluation turned out to be the ultimate combination

to examine the research questions. The possible promising effect of COPCA is in line with the

178 179

results from the systematic reviews which indicated that general developmental programmes are

the most promising to influence developmental outcome in infants with or at high risk for devel-

opmental motor disorders.

In the next years the multicenter trial Learn to Move 0-2 years will provide more knowledge about

the effect of early intervention on the developmental outcome of infants at high risk for cerebral

palsy in general and the COPCA programme in particular.

S a M E N Vat t I N G

Jonge kinderen met een verhoogd risico op het krijgen van een motorische ontwikkelingsstoornis,

zoals cerebrale parese (CP) of developmental coordination disorder, komen in aanmerking voor

vroegtijdige interventie. De term vroegtijdige interventie omvat een scala aan multidisciplinaire

therapeutische mogelijkheden die zich richten op de ontwikkeling en opvoeding van kinderen

tussen nul en vijf jaar. In Nederland worden kinderen met een verhoogd risico op het krijgen van

een motorische ontwikkelingsstoornis vaak verwezen naar de kinderfysiotherapeut. In dit proef-

schrift is vroegtijdige interventie afgebakend tot kinderfysiotherapie voor kinderen jonger dan

de gecorrigeerde leeftijd van 18 maanden. De introductie (Hoofdstuk 1) geeft een overzicht van de

belangrijkste ontwikkelingen binnen de kinderfysiotherapie in Nederland. Kinderfysiotherapie is

een discipline die zich de afgelopen decennia sterk heeft ontwikkeld. Er zijn een aantal belangrijke

verschuivingen opgetreden. De nadruk ligt tegenwoordig op functionaliteit in plaats van normale

bewegingspatronen. Ook wordt het gezin actief betrokken bij het therapeutisch proces en neemt

het kind actief deel aan zijn of haar eigen therapie.

Het eerste doel van dit proefschrift was het in kaart brengen van de bestaande effecten van

vroegtijdige fysiotherapeutische interventie op de motorische en cognitieve ontwikkeling van

kinderen die jonger zijn dan 18 maanden en een verhoogd risico hebben op het krijgen van een

motorische ontwikkelingsstoornis (Hoofdstuk 3 en 4). Uit de beide systematische literatuurstudies

blijkt dat het effect van interventies die zich primair richten op het verbeteren van de motorische

ontwikkeling onduidelijk is. Het type interventie dat mogelijk effect heeft, verschilt voor kinderen

op de preterme leeftijd en kinderen die tenminste de à terme leeftijd bereikt hebben. Voor de à

terme leeftijd lijken kinderen het meest baat te hebben bij interventies waarin de situatie van de

baarmoeder wordt nagebootst. Na de à terme leeftijd hebben programma’s die zich richten op de

algemene ontwikkeling van het kind en waarin ouders leren hoe ze de ontwikkeling van hun kind

kunnen stimuleren de meeste potentie. De traditionele benaderingen, zoals NeuroDevelopmental

Treatment en behandeling volgens de methode Vojta, hebben geen effect op de ontwikkeling van

kinderen met een verhoogd risico op het krijgen van een motorische ontwikkelingsstoornis.

De resultaten van beide literatuurstudies laten zien dat interventie programma’s, die starten

na de à terme leeftijd en zich richten op het stimuleren van zelf geproduceerd motorisch gedrag,

de motorische en cognitieve ontwikkeling positief kunnen beïnvloeden. Deze kennis heeft

bijgedragen aan de ontwikkeling van een nieuwe vorm van kinderfysiotherapeutische interven-

tie genaamd COPCA (Coping with and Caring for infants with special needs - a family-centered

programme). COPCA is gericht op de relaties en interacties binnen het gezin. Het programma

is gebaseerd op nieuwe inzichten op het gebied van pedagogiek en gezinsgerichte zorg en op de

principes van de Neuronale Groep Selectie Theorie.

180 181

Het tweede doel van dit proefschrift was het evalueren van het effect van COPCA op de ontwikke-

ling van kinderen met een verhoogd risico op het krijgen van een motorische ontwikkelingsstoor-

nis. Aan het Vroegtijdig Interventie Project (VIP; Hoofdstuk 2) hebben 46 kinderen deelgenomen.

De kinderen zijn via blokrandomisatie verdeeld over de experimentele COPCA groep (21 kinderen)

en de controlegroep (25 kinderen) die de traditionele vorm van kinderfysiotherapeutische begelei-

ding (TIP) op indicatie van de kinderarts kreeg. De interventieperiode duurde van 3 tot 6 maan-

den. De ontwikkeling van de kinderen is gemeten op de gecorrigeerde leeftijd van 3, 4, 5, 6 en 18

maanden aan de hand van meetinstrumenten die zich richten op de neuromotore ontwikkeling,

het psychosociaal functioneren en de neurofysiologische parameters van de houdingsregulatie.

Uit de analyses kwam naar voren dat er op groepsniveau (RCT) weinig verschillen waren

tussen de COPCA en de TIP groep (Hoofdstuk 7), behalve in de cognitieve ontwikkeling. Tien kin-

deren hadden een CP ontwikkeld op de gecorrigeerde leeftijd van 18 maanden. Het merendeel van

de kinderen (15 COPCA, 16 TIP) liet de complexe vorm van minor neurological dysfunction zien,

waardoor ze op de schoolleeftijd meer risico lopen op leer- en gedragsproblemen. Twee interes-

sante resultaten kwamen uit het RCT naar boven. Ten eerste bestond er een interactie tussen het

opleidingsniveau van de moeder en het type interventie met de verandering in de cognitieve uit-

komst tussen 6 en 18 maanden. Dit betekende dat kinderen van moeders met een lagere opleiding

uit de COPCA groep minder achteruitgingen in hun MDI score dan andere kinderen. Dit sugge-

reert dat moeders met een lager opleidingsniveau meer profiteren van het coachen door een the-

rapeut die de COPCA methode gebruikt. Ten tweede viel op dat de neurologische uitkomst in de

COPCA en de TIP groep vergelijkbaar was. Dit is interessant gezien de tegenovergestelde kijk van

TIP en COPCA op neurologische parameters zoals spierspanning en afwijkende bewegingen. In

TIP vormen tonus beïnvloedende strategieën een belangrijke rol in de fysiotherapeutische acties,

terwijl COPCA hier geen aandacht aan besteed. Oftewel, fysiotherapeutische technieken gericht

op het beïnvloeden van de spierspanning en het faciliteren van ‘normale’ bewegingspatronen lij-

ken geen effect te hebben op de neurologische ontwikkeling. In Hoofdstuk 8 staan een aantal aan-

vullende resultaten van meetinstrumenten uit het VIP project beschreven die niet gepubliceerd

zijn. Andere uitkomsten worden momenteel geanalyseerd, zoals de motorische ontwikkeling in

termen van de Infant Motor Profile, parameters van de houdingsregulatie en video opnames van

activiteiten in het dagelijks leven (baden en spelen).

Het derde doel van dit proefschrift was de evaluatie van de toepasbaarheid van COPCA in de da-

gelijkse praktijk. Daarvoor zijn video opnames gemaakt van fysiotherapeutische behandelingen op

4 en 6 maanden. Om de inhoud van de fysiotherapeutische behandeling te kunnen analyseren, is

eerst een gestandaardiseerd observatie protocol ontwikkeld (Hoofdstuk 5). Dit protocol is opge-

bouwd uit de belangrijkste elementen en kern items van kinderfysiotherapeutische interventie.

Fysiotherapeutische handelingen worden ingedeeld in 8 categorieën: A) Mate van betrokkenheid

van het gezin en opvoedingselementen gericht op het kind/gezin, B) Communicatie, C) Facilitatie

technieken, D) Sensorische stimulatie, E) Passief bewegen, F) Zelf geproduceerd motorisch gedrag

zonder tussenkomst van de fysiotherapeut of ouder G) Uitgelokt zelf geproduceerd motorisch

gedrag dat wordt voortgezet door het kind, H) Uitgelokt zelf geproduceerd motorisch gedrag dat

overgaat in handling. Analyses lieten zien dat het protocol voldoet aan belangrijke psychome-

trische eisen zoals een voldoende inter- en intrabeoordelaarsbetrouwbaarheid en dat bijna alle

fysiotherapeutische handelingen gescoord kunnen worden in de categorieën van het protocol. Het

observatieprotocol kan gebruikt worden als instrument om de heterogeniteit in de fysiotherapie

te meten. Het geeft een gedetailleerd overzicht van de handelingen die een kinderfysiotherapeut

toepast binnen zijn of haar behandeling en houdt tegelijkertijd rekening met verschillende theore-

tische referentiekaders. Uit de studies waarin het observatieprotocol toegepast is (Hoofdstuk 5-7),

kunnen vier conclusies worden getrokken:

1) Traditionele kinderfysiotherapeutische begeleiding wordt gekenmerkt door een zeer heterogene

toepassing. Dit wordt mogelijk veroorzaakt door het integreren van technieken uit verschillende

stromingen binnen één behandeling.

2) Er bestaat een discrepantie tussen de werkelijke fysiotherapeutische handelingen en de inter-

pretatie van de kinderfysiotherapeut over de handelingen die hij/zij toepast.

3) De inhoud van COPCA en TIP is zeer verschillend (Hoofdstuk 6). Dit suggereert dat het mogelijk

is om COPCA te implementeren in de dagelijkse praktijk, mits het gegeven wordt door fysiothe-

rapeuten die een formele training in de COPCA methode gevolgd hebben. De verschillen tussen

COPCA en TIP corresponderen met de theoretische kaders van beide interventieprogramma’s.

Er bestaan verschillen in de rol van de ouders en de benadering van de familie, in de opvoe-

dingselementen gericht op het kind en in activiteiten die zich richten op het stimuleren van de

sensomotorische ontwikkeling van het kind.

4) De inhoud van de behandeling speelt wel degelijk een rol bij de ontwikkeling van kinderen met

een hoog risico op een motorische ontwikkelingsstoornis. Een aantal aspecten van de COPCA

interventie , namelijk de mate van betrokkenheid van de familie en opvoedingselementen

gericht op het kind/gezin (met name coaching), het uitlokken van zelf geproduceerd motorisch

gedrag dat wordt voortgezet door het kind met veel variatie en het stimuleren van motorisch ge-

drag op de grens van de mogelijkheden die een kind heeft, zijn geassocieerd met een betere ont-

wikkelingsuitkomst. Het gebruik van facilitatie technieken, zoals handling, was geassocieerd

met een slechtere uitkomst op de functionele vaardigheden schaal van de PEDI. Sensorische

stimulatie en passief bewegen waren op de korte termijn geassocieerd met een betere cognitieve

ontwikkeling, maar dit verband was niet meer aanwezig op 18 maanden. Het is belangrijk om te

beseffen dat de resultaten van deze studie voorzichtig geïnterpreteerd dienen te worden aange-

zien het bestaan van een associatie niet automatisch betekent dat er een causale relatie bestaat

tussen de inhoud en de uitkomstmaat. Associaties tussen fysiotherapeutische handelingen en

ontwikkeling zijn onvermijdelijk gecontamineerd met de uitgangssituatie van het kind. Om dit

probleem te ondervangen hebben we gebruik gemaakt van multivariate statistiek, maar er is

meer onderzoek nodig om de invloed van de inhoud van fysiotherapeutische interventie op de

ontwikkeling vast te kunnen stellen.

182

In Hoofdstuk 9 wordt een overzicht gegeven van de belangrijkste bevindingen uit dit proefschrift.

Alhoewel de verschillen tussen de COPCA en de TIP groep op groepsniveau klein zijn, lijkt het

erop dat COPCA mogelijk de ontwikkeling van kinderen die een verhoogd risico hebben op het

ontwikkelen van een motorische ontwikkelings-stoornis positief kan beïnvloeden. Deze gedachte

wordt m.n. ondersteund door de bevinding dat een aantal kernitems van COPCA, namelijk

coachen, variëren, uitdagen en exploreren, geassocieerd zijn met een betere ontwikkeling. We

concludeerden dan ook dat het uitbreiden van de RCT met een procesevaluatie de mogelijkheid

schepte om de onderzoeksvragen goed te kunnen beantwoorden. Het mogelijke positieve effect

van COPCA komt overeen met de resultaten van de systematische literatuurstudies waaruit

blijkt dat vroegtijdige interventie programma’s, die zich richten op de algehele ontwikkeling

van het kind en waarin ouders leren hoe ze de ontwikkeling van hun kind kunnen stimuleren,

de ontwikkeling van kinderen met een verhoogd risico op het krijgen van een motorische

ontwikkelingsstoornis positief kunnen beïnvloeden.

In de komende jaren zal het onderzoek Learn to Move 0-2 jaar meer inzicht verschaffen in het ef-

fect van vroegtijdige interventie op de ontwikkeling van kinderen met een hoog risico op cerebrale

parese in het algemeen en in het COPCA programma in het bijzonder.

184 185

Da N K WO O r DEindelijk ligt het er dan, mijn proefschrift. Natuurlijk heb ik dit niet alleen gedaan en hebben veel

mensen een bijdrage geleverd aan het tot stand komen van dit proefschrift, direct dan wel indirect.

Ik zou graag op deze plek graag iedereen persoonlijk willen bedanken, maar het is onmogelijk om

iedereen bij naam te noemen. Daarom wil ik graag, voor ik enkele mensen persoonlijk ga bedanken,

alle teamgenoten, toeschouwers en ontspanners, ontzettend bedanken! Zonder jullie steun en aan-

moediging was het nooit gelukt!

Allereerst wil ik de 46 kinderen en hun ouders die hebben deelgenomen aan het VIP project ontzet-

tend bedanken voor hun bereidwilligheid, hun inzet en hun vertrouwen om ons een kijkje te geven

in hun gezin.

Prof. dr. M. Hadders-Algra, beste Mijna, de draaiende motor van het instituut voor Ontwikkelings-

neurologie en mijn eerste en enige promotor. Dankzij jouw enorme betrokkenheid en enthousiasme

is dit proefschrift mede tot stand gekomen. Ik heb in de afgelopen jaren enorm veel van je geleerd

en wil je bedanken voor je vertrouwen dat het met mijn proefschrift wel een keer goed zou komen

(al heb je daar misschien tussendoor wel eens – net als ikzelf – aan getwijfeld). Ik heb me vaak afge-

vraagd uit hoeveel uur jouw dag eigenlijk bestond. Of ik nu ’s avonds of in het weekend een e-mail

stuurde, altijd was daar een bijzonder snelle reactie op mijn vragen en had je de stukken van com-

mentaar en correcties voorzien.

Tineke Dirks, ook al ben je niet officieel promotor, toch voelt het wel een beetje zo. Wat heb ik veel

van jou geleerd over de inhoud van de kinderfysiotherapie. NDT, Vojta, en als klap op de vuurpijl

COPCA. Uren heb je met het popje voor me gezeten om me alle handelingen die een kinderfysiothe-

rapeut met een baby kan uitvoeren, uit te leggen. En dat zijn er nogal wat! Onze oneindige discus-

sies over de inhoud van het observatieprotocol hebben toch al mooi tot drie artikelen geleid.

Prof. Dr. J.G. Becher, Prof. Dr. O.F. Brouwer en Prof. Dr. P.J.M. Helders wil ik bedanken voor het

beoordelen van mijn proefschrift.

Prof.Dr. A.F. Bos en drs. L. van Doormaal wil ik hartelijk bedanken voor hun bijdrage aan het VIP

project. Arie, daarnaast wil ik je bedanken voor je hulp bij het samenstellen van de onderzoeksgroep,

onze gezamenlijke artikelen en het interpreteren van de schedelecho’s.

Kune Blaauw-Schierbeek, Ellen Janssen, Peter Logtenberg en drs. Wilma Renkema, COPCA coaches

van het eerste uur. Jullie hebben een zeer belangrijke rol in het onderzoek gespeeld en geweldig

werk geleverd. Het volledig ombouwen van je dagelijkse routine tot het compleet beheersen van een

nieuwe methode hebben jullie goed onder de knie gekregen. Mijn complimenten!

Beste collega’s van de ontwikkelingsneurologie: Lieke van Baalen, Hylco Bouwstra, Leo van Eykern,

Saskia van Goor, Jolanda van der Heide, Kirsten Heineman, Corina de Jong, Marjolein Jongbloed-

Pereboom, Karin Middelburg, Lieke Peters, Michiel Schrier en Loes de Weerd. Jullie wil ik bedanken

voor al jullie steun, collegialiteit, hulp bij het onderzoek, het filmen, de EMG-metingen en jullie be-

reidheid een luisterend oor te bieden als het weer eens niet liep met het onderzoek of het schrijven.

De studenten die ik mocht begeleiden bij hun doctoraalscriptie of wetenschappelijke stage hebben

mij erg geholpen met het verzamelen en verwerken van de resultaten. Renske van Abbema, Eveline

Nieuwenhuis en Lily Hulshof, jullie hebben een grote bres geslagen in de enorme hoop onderzoeks-

gegevens en van jullie frisse blik op het onderzoek heb ik veel geleerd.

Victorine de Graaf-Peters, mijn raamloze kamergenoot en nu nog steeds vriendin. Je bent met ruime af-

stand de meest praatgrage collega, maar het was altijd weer gezellig om even het weekend of resultaten

door te nemen. Toen je weg was, vond ik het opeens maar stil. Zelfs de sneeuw heb ik gemist. Gelukkig

kun je nu af en toe wat sneeuw vanuit Amersfoort sturen! Geweldig dat je mijn paranimf wilt zijn.

Joke de Boer, wat begon als bovenbuurvrouw en volleybalmaatje is inmiddels uitgegroeid tot veel ge-

zellige muziek-, eet-, en filmavondjes met zijn vieren. Misschien moeten we de frequentie maar weer

eens verhogen… Ik ben enorm blij dat je mijn paranimf wilt zijn. Mark, jou wil ik bedanken voor al je

hulp bij het vormgeven, lay-outen en alle drukadviezen. Ik ben enorm trots op het resultaat en zal je

bij iedereen die een grafisch ontwerper nodig heeft van harte aanbevelen.

Met mijn verhuizing naar het KNGF kreeg ik er een aantal andere geïnteresseerde collega’s voor terug.

Katja Aberson, Marion Bosga, Jeroen Broekhoff, Karin Heyl, en Charlotte Hilbers, het Heerenveen dream-

team! Nou dames en heer, het zit er eindelijk op. Bedankt voor al jullie interesse en veel plezier met lezen…

Lieve pap en mam, altijd staan jullie voor mij klaar. Ik wil jullie dan ook bijzonder graag bedanken

voor de enorme steun en het onvoorwaardelijke vertrouwen dat jullie mij altijd gegeven hebben.

Dan nu speciaal iets voor mijn grote knul Yonah. Lieve, lieve eigenwijze kerel. Je begreep nog niet

altijd goed waar mamma nou al die tijd druk mee was. “Mamma, gaat jij nu weer werken op de kap-

joeter…?’. Maar Yonah, nu is het af en kan ik ook mee fietsen en koeien zoeken in het park op zon-

dagmiddag. En over een paar maanden gaan we met z’n vieren en ben jij de grote broer!

Lieve Levy, als ik moet opsommen waarvoor ik jou wil bedanken, ben ik wel even bezig. Wat een

luxe om jou als partner te hebben. Je hebt me enorm geholpen om structuur te krijgen in de enorme

data-berg. Van jouw fantastische Excel-formules heb ik zeven jaar plezier gehad! Gelukkig kan ik van

jou nog veel langer genieten. Laten we er iets moois van maken, niet alleen met z’n viertjes, maar

ook met elkaar.

186 187

a B O U t t H E aU t H O r

Cornill Blauw-Hospers werd geboren op 8 januari 1978 te Groningen. Na het behalen van haar

VWO diploma aan het Ubbo Emmius Lyceum te Stadskanaal in 1996 startte zij met de opleiding

Bewegingswetenschappen aan de Rijksuniversiteit Groningen. In 2000 studeerde zij af met als

specialisatie Sport en Lichamelijke opvoeding. Haar afstudeerproject betrof de evaluatie van het

effect van fysiotherapie voor kinderen met motorische problemen. Na haar afstuderen werkte

zij enige tijd als onderzoeker bij het Centrum voor Revalidatie Beatrixoord van het Universitair

Medisch Centrum Groningen. Na een verhuizing naar Sneek zette zij haar onderzoeksloopbaan

voort in Groningen waar zij op 1 maart 2003 werd aangesteld als AIO bij het Instituut voor Ontwik-

kelingsneurologie van het UMCG. Zij deed daar onderzoek naar de effecten van vroegtijdige fysio-

therapeutische interventie voor jonge kinderen met een hoog risico op een motorische ontwik-

kelingsstoornis. De resultaten van haar studie staan beschreven in deze dissertatie. Vanaf september

2008 is zij werkzaam bij het Koninklijk Nederlands Genootschap Fysiotherapie te Heerenveen.

Hospers CH, De Graaf-Peters VB, Hadders-Algra M. Het vroegtijdige interventie project in Groningen. Ned Tijdschr Kinderfysiother 2004; 16, nr 40:14-16.

Hadders-Algra M, Dirks T, Blauw-Hospers C, de Graaf-Peters V. The Kozijavkin method: giving parents false hope? Lancet 2005;365:842. Blauw-Hospers CH, Hadders-Algra M. Fact or fiction: a systematic review on the effects of early intervention on motor development. Dev Med Child Neurol 2005;47:421-432.

De Graaf-Peters VB, Blauw-Hospers CH, Dirks T, Bakker J, Bos AF, Hadders-Algra M. Development of postural control in typically developing children and children with cerebral palsy: possibilities for intervention? Neurosci Biobehav Rev 2007;31:1191-1200.

Blauw-Hospers CH, de Graaf-Peters VB, Dirks T, Bos AF, Hadders-Algra M. Does early intervention in infants at high risk for a developmental motor disorder improve motor and cognitive development? Neurosci Biobehav Rev 2007;31:1201-1212.

Blauw-Hospers CH, Dirks JF, Hulshof LJ, Hadders-Algra M. Opening the Black-Box: development of a quantitative tool to assess physical therapy contents in infancy. Pediatr Phys Ther 2010;22:189-198.

Blauw-Hospers CH, Dirks T, Hulshof LJ, Bos AF, Hadders-Algra M. A Randomized Controlled Trial on pediatric physical therapy in infancy: from nightmare to dream. Submitted for publication.

Dirks T, Blauw-Hospers CH, Hulshof LJ, Hadders-Algra M. Differences between a Family Centered Program “Coping and Caring for Infants with Special Needs” and Infant Treatment based on Principles of NeuroDevelopmental Treatment. Submitted for publication.

Hielkema T, Blauw-Hospers CH, Dirks T, Drijver-Messelink M, Bos AF, Hadders-Algra M. Does physiotherapeutical intervention affect motor outcome in high risk infants? A combined approach of RCT and process evaluation. Submitted for publication.

L I S t O F P U B L I C at I ON S

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Observation protocol: classification of pediatric physical therapy ( PT) actions

The PT actions are classified into 8 main categories which contain various subcategories (second level of observation). For each specific action one or more examples of concrete PT or caregiver actions are provided (third level of observation)

a. Family involvement and educational actionsThe extent to which the family is involved in the treatment of the infant and the extent of guidance, in-terference, coaching or training by the physical thera-pist or the caregiver during the treatment session.

a.1 Family members participating in guidance infant*: Family members involved in the treatment session.Examples of concrete actions:- Mother present only - Father present only - Both caregiver but no other family members present - In addition to parent(s) also other family members

present

a.2 role of caregiver*: The extent to which the family is involved in the treatment.

Examples of concrete actions:- PT performs treatment by means of handling

techniques- Caregiver and PT act together in handling tech-

niques, PT performs the treatment (hands on) while the caregiver guides the attention of the infant.

- Caregiver performs handling techniques thereby controlling the infant’s actions. The PT instructs the caregiver how to handle

- Caregiver and PT act together (hands off), caregiver is playing with the child and may provide the infant with minimal support but leaves the infant always with ample opportunities for exploration. PT observes the caregiver-infant relationship and may give hints.

- Caregiver is playing with the infant (hands off)

and leaves the infant with ample opportunities for exploration

a.3 Infant dressing*: The way in which the infant is dressed during the treatment session.Examples of dressing: - Infant is dressed - Infant is partially dressed - Infant is undressed (wearing underwear only)

a.4 Educational actions A.4.1 The extent of interference by the PT or the caregiver with infant’s activities Examples of concrete actions: - PT or caregiver interrupts activities of the infant. - PT or caregiver corrects when infant fails. - PT or caregiver assists when infant has difficul-

ties to perform an action A.4.2 The extent of guidance the infant by the PT.Examples of concrete actions: - PT trains motor performance at the level which is

relatively too easy for the infant. - PT provides excessive postural support or assistance. - PT trains motor performances at the level which is

too difficult for the infant.

A.4.3. Caregiver training All actions during which the PT instructs caregivers how to handle the infant with the aim that caregiv-ers can continue treatment strategies during daily life activities and/or in the home environment.Examples of concrete actions: - PT demonstrates therapeutic handling action to

caregiver. - PT demonstrates action to caregiver, variable op-

tions provided.- PT practices with caregivers teaching them how to con-

tinue some of handling techniques in daily life at home.

A.4.4 Caregiver coachingAll actions during which the PT coaches the caregiv-er. Coaching aims to empower caregivers so that they can made their own educational decisions during daily care activities in the home environment.

a P P E N DI x Examples of concrete actions:- Caregiver patiently observes the infant actions- Caregiver challenges motor performances just at

the verge of infant’s abilities.- Caregiver provides as little postural support as pos-

sible – challenges postural behavior of the infant- Caregiver tries to evoke pleasure in the infantB. Communication All communication between the PT and the caregiver that is related to the guidance of infant and family

B.1 Information exchange: All communication during which information is exchanged regarding experiences, worries and the role of the PT.Examples of concrete actions:- PT provides opportunity for caregivers to tell about

experiences related to NICU stay, to express worries and concerns about infant and family matters.

- PT provides information about role of PT and caregiver.

- PT asks whether specific problems exist.

B.2 Contents of information: All communication that explains the background of the treatment strategies, including developmental education.Examples of concrete actions: - PT explains handling in terms of typical movement

patterns, typical development, posture, muscle tone, asymmetry/symmetry and hand placing.

- PT explains the need of, variation, minimal support, exploration, trial and error, challenge, patience.

- PT [explains the need to explore communication.- PT discusses the application of intervention strate-

gies into daily routines in terms of handling.- PT discusses the application of the intervention

into daily routines in terms of variation, explora-tion, motor challenge

B.3 Instruct: All communication in which the caregiver is given assignments or hints regarding treatment strategies.

Examples of concrete actions:- PT assigns, gives advice what to do. - PT gives hints, provides a suggestion or clue in

a very indirect way so that caregivers feel free to explore ample variable opportunities.

B.4 Provide Feedback: All communication in which the treatment or the performances of infant and caregiver are evaluated.Examples of concrete actions:- PT tells the caregiver what went right/wrong. - PT evaluates the procedure. - PT asks and listens to the opinion of the caregiver.B.5 Impart knowledge: All communication that provides the caregiver with knowledge about the therapeutic actions that are performed.Examples of concrete actions:- PT asks about performance action. - PT explains the ins and outs of an action. - PT asks about understanding. - PT asks about ability of caregiver to perform an ac-

tion and listen to caregivers comments on actions.

C. Facilitation techniques. All therapeutic hands on actions of the physical therapist or caregiver aiming at guidance of the movement or maintenance of the infant’s posture by gently placing the hands on specific parts of the infant’s body, thus providing the infant with sensori-motor experience and controlling movement output.

C1. Handling:Specific hands-on techniques to give the infant sensorimotor experience to improve the quality and repertoire of the infant’s movements.Examples of concrete PT/caregiver actions:- In supine position or in sitting. Shoulders function

as key point: handling hands guide shoulders of the infant in protraction to control tone and to facili-tate hand-hand contact and symmetry.

- In supine position. Proximal or distal leg functions as key point: the infant’s hip is passively brought in semi-flexion while adducting the leg across the midline to facilitate head righting and rolling.

190 191

- In supine position. Pelvis functions as key point: the infant’s pelvis is slightly lifted to elongate the extensor muscles of the trunk and to control tone; in this way hand-foot contact and symmetry are facilitated

- In prone position. Shoulder functions as key point, the arms are placed in puppy position to facilitate head righting, midline orientation and body-alignment.

- In sitting. Shoulder functions as key point: the shoulders are moved alternately forwards and backwards to dissociate and facilitate independent arm movements.

C.2 Pressure techniques: All handling techniques that produce intermittent pressure to stimulate and gain control over muscle tone, posture and movement. Examples of concrete PT /caregiver actions: - In sitting: intermittent downward pressure on

shoulders in the direction of the pelvis to facilitate extension of the trunk

- In sitting: slight intermittent pressure movements on abdominal region in direction of the sacrum to facilitate contraction of the ventral muscles.

C.3 transition: All handling techniques that result in the change of position of the infant.Examples of concrete PT /caregiver actions: - From supine to side, from supine to prone, from

supine to sit, from side to sit, from prone to supine, from sit to supine, etc.

C.4 Support devices: All handling techniques which use additional devices to support the infant.Examples of concrete / additional devices: - Bolster or ball- Supporting sling.

D. Sensory experience All tactile and vestibular stimulation given to the infant during treatment - without the aim of facilita-tion, tapping or passive motor experience - offered to

the infant to provide him/her with the perception of body awareness.Examples of concrete PT /caregiver actions: - Touching skin with toy.- Tickling. - Tapping on muscles

E. Passive motor experienceAll handling techniques induced by the PT or the caregiver in which no activity of the infant is re-quired in the performance of the actions.Examples of concrete PT /caregiver actions:- Passive movements of arms. - Repetitive movements of the upper arm towards

(frontal) support surface.- Passive rocking, small sideways movements.

F. Self-produced motor behavior, no interference with Pt /caregiver All actions during which the infant is given ample opportunities to explore toys or other aspects of the environment or his body without interference of PT or caregiver.Examples of concrete PT /caregiver actions:- Placing an infant activity play center over the infant

and let the infant explore the effect of movements or arms, hands, legs and feet.

- Infant is given opportunity for spontaneous explo-ration with or without toy.

- Postural challenges, infant spontaneously explores postural capacities.

G. Infant is challenged to produce motor behavior by him/herself; infant is allowed to continue activ-ity by her/himselfAll actions in which the infant is challenged by toys or face of PT or caregiver to experience a variety of motor activity, that is continued by the infant her/himself.

G.1 Little variation: All actions in which the infant is challenged by toys or face of PT or caregiver to explore one strategy to reach and grasp, to control posture, to roll, etc.

G.2 Large variation: All actions in which the infant is challenged by toys or face of PT or caregiver the infant is challenged to explore multiple strategies to reach and grasp, to control posture, to roll, etc.

H. Infant is challenged to produce motor behavior by him/herself; activity flows over into therapeutic handling All actions in which the infant is challenged by toys or face of PT or caregiver to experience a variety of motor activity, that is followed by a handling technique.

H.1 Little variation: All actions in which the infant is challenged by toys or face of physical therapist or caregiver to explore one strategy to reach and grasp, to control posture, to roll, etc.

H.2 Large variation: All actions in which the infant is challenged by toys or face of physical therapist or caregiver the infant is challenged to explore multiple strategies to reach and grasp, to control posture, to roll, etc.

I. Not specified All time during the treatment session that can not be classified into the eight defined categories.Examples: - Comforting the infant. - Change the treatment situation.

Postural support in prone, side and sitting position: No postural support: PT or caregiver leaves it to the infant to adjust pos-ture independently. “Hands off” Minimal postural support: PT or caregiver provides as little support as possible in order to challenge postural behavior of the infantExample of concrete action:PT or caregiver challenges motor performance just at the verge of the infant’s abilities, i.e. the infant has to ‘work’ to maintain balance

Clear postural support: PT or caregiver provides support on multiple parts of body or the trunk. Minimal active involvement of the infant to adjust posture is required.Example of concrete action:- PT or caregiver provides support at the neck/shoul-

der girdle and/or upper part of the trunk Full postural support: PT or caregiver supports all parts of the body of the infant that play a role in postural adjustments. No active involvement of the infant is required.

* Independent variable: the value of this variable is supposed not to change during the course of an observation. It gives the observer the opportunity to summarize briefly important characteristics of the observation